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Adaptation to Inflammatory Bowel Disease by Lawrence Matini Thesis submitted for the award of Doctor of Philosophy January 2017 School of Psychology Faculty of Health and Medical Sciences University of Surrey Declaration of originality This thesis and the work to which it refers are the results of my own efforts. Any ideas, data, images or text resulting from the work of others (whether published or unpublished) are fully identified as such within the work and attributed to their originator in the text. This thesis has not been submitted in whole or in part for any other academic degree or professional qualification. I agree that the University has the right to submit my work to the plagiarism detection service TurnitinUK for originality checks. Whether or not drafts have been soassessed, the University reserves the right to acquire an electronic version of the final document (as submitted) for assessment as above. Signed: Date: Abstract Inflammatory bowel disease (IBD) is a term used to describe two chronic diseases of the gastrointestinal tract: Ulcerative Colitis (UC) and Crohn's Disease (CD). Although the efficacy of treatment is continuously improving, Quality of Life (QoL) in this illness population remains low with many patients suffering from psychological and psychiatric comorbidities. Psychological interventions aimed at improving outcomes in these patients have largely demonstrated little improvement. This thesis argues that this may be the result of poor understanding of the experience of living with this condition with too little focus on the adaptation of the patient to their illness. This thesis aimed to address this gap in the literature through four empirical studies. Firstly, Study 1 used a qualitative design to (n = 22) to explore the lived experiences of patients with IBD and to conceptualise adaptation to IBD. The results highlighted the importance of making sense of the illness and the impact and feelings associated with the illness. This was transcended by a notion of uncertainty which was resolved by employing coping mechanisms to restore equilibrium between their identity before and after diagnosis, resulting in a 'new normal'. Study 2 then employed a crosssectional design (n = 307) to develop a new measure of adaptation to IBD (the A-IBD) which after psychometric analysis revealed four subscales including person identity, patient identity, acceptance and expectations. This study also explored the degree of association of the A-IBD with existing measures of sense making (BIPQ) and QoL (IBDQ), to assess the ability of the A-IBD in predicting QoL and ascertain whether it could predict QoL over and above sense making. The results suggested the A-IBD was not synonymous with these constructs and had utility as a predictor of QoL even when accounting for the predictive ability of the BIPQ. Finally, Study 3 used a combination of qualitative and quantitative design (n = 16). Patients scoring in the top and bottom 25% of the A-IBD from Study 2 completed the measure again to assess the dynamic nature of adaptation and were interviewed about the factors that either encouraged or inhibited their degree of adaptation. This analysis revealed that adaptation is indeed dynamic, and that antecedents of adaptation include 'engagement', 'resilience' and certain 'contingencies' including disease and social factors. Overall, the findings from this thesis indicate that the treatment of IBD must be approached in a biopsychosocial manner, that adaptation can be measured effectively with the new tool and that adaptation, with an emphasis on the notion of person, not patient, predicts quality of life. Acknowledgements Firstly, I would like to extend my utmost gratitude to my supervisor, Professor Jane Ogden, who has supported and encouraged me every step of the way over the past three years. Your understanding throughout the toughest point in my life health-wise gave me the time and space I needed to re-focus and see this PhD through to the end. I couldn't have wished for a better person to be my supervisor, so thank you. I would also like to thank Dr. Deborah Cooke for her support and guidance as well as the numerous other staff and students of the University of Surrey School of Psychology that have assisted me along this journey. I must also thank the School of Psychology for selecting me to be a recipient of the Russell Wicks memorial fund which was of great help in seeing me through the final year of this PhD. I of course also owe a huge thanks to all the individuals who took part in my research. The willingness of those who gave up their time and shared their personal stories with me was staggering and I am forever grateful. I hope this PhD contributes to an increased awareness and better management of IBD and thus goes some way towards paying back all that were involved. Finally, I would like to thank my family and closest friends. My Mum, Dad and Brother have been unwavering in their support ever since I expressed my desire to one day complete a PhD a number of years ago. Through the toughest of times they continued to stand by me and help me realise this dream. I'm particularly glad my Dad can now tell everyone about how his son is a doctor without me having to stop him. Table of Contents Chapter One Literature Review 1 1.1 Definition of IBD 1.2 Prevalence of IBD 1.3 Causes of IBD 1.4 Consequences of IBD 1 5 10 15 15 21 25 26 26 30 32 42 43 46 1.4.1 Physiological consequences 1.4.2 Psychological consequences 1.5 IBD as a chronic condition 1.6 The process of having a chronic condition 1.6.1 Coping 1.6.2 Criticisms of the coping literature 1.6.3 Sense making 1.6.4 Criticism of the SRM and CAT 1.7 Outcomes of a chronic condition 1.8 The working model of adjustment to chronic illness – integrating both processes and outcomes 1.9 The central role of adaptation 1.10 Process, outcomes and adaptation in IBD 1.11 Overview of aims for thesis 49 50 54 Chapter Two Study 1: Patients’ experiences of living with IBD: a qualitative study 57 2.1 Overview 2.2 Introduction 2.3 Method 57 57 60 60 60 63 63 2.3.1 Design 2.3.2. Sample 2.3.3. Procedure 2.3.4 Interview schedule 2.3.5 Data analysis 2.4 Results 2.5 Discussion 2.5.1 Summary of findings 64 76 76 2.5.2 Links to literature 2.5.3 In summary 2.5.4 Methodological limitations 2.5.5 Conclusion 77 78 78 80 Chapter Three Study 2: The development of a new measure to assess adaptation in patients with IBD and its utility in predicting QoL: a cross-sectional study 81 3.1 Overview 3.2 Introduction 81 82 82 85 3.2.1 Literature review 3.2.2. Steps involved in development and validation of the adaptation to IBD questionnaire 3.3 Method 3.3.1 Design 3.3.2 Sample 3.3.3 Procedure 3.3.4 Measure 3.4 Results 3.4.1 Conceptualisation 3.4.2 Operationalisation 3.4.3 Assessment of the scale’s psychometric properties 3.4.4 Reliability assessment 3.4.5 Final scale with response options and instructions 3.4.6 Association of the A-IBD with existing measures of sense making and QoL 3.4.7 Using the A-IBD subscales to predict QoL 3.4.8 Adaptation formula 3.4.9 Does the A-IBD predict QoL over and above the BIPQ? 3.5 Discussion 3.5.1 Summary of findings 3.5.2 Links to literature 3.5.3 In summary 3.5.4 Methodological limitations 3.5.5 Conclusion 87 87 87 87 88 89 89 90 91 97 97 99 102 111 111 113 113 115 117 119 122 Chapter Four Study 3: Patients’ explanation for their degree of adaptation: a qualitative study with a quantitative component 123 4.1 Overview 4.2 Introduction 4.3 Method 123 124 126 126 126 127 127 129 129 130 130 148 148 4.3.1 Design 4.3.2 Sample 4.3.3 Classifying degree of adaptation 4.3.4 Change in adaptation 4.3.5 Procedure 4.3.6 Interview schedule 4.3.7 Data analysis 4.4 Results 4.5 Discussion 4.5.1 Summary of findings- thematic analysis 4.5.2 Links to literature - thematic analysis 4.5.3 Summary of findings – adaptation - state or trait? 4.5.4 Links to literature - adaptation – state or trait? 4.5.5 In summary 4.5.6 Methodological limitations 4.5.7 Conclusion 148 150 150 151 152 153 Chapter Five General Discussion 154 5.1 Overview 5.2 Summary of findings 5.3 Story of the thesis – What is adaptation? 5.4 Methodological limitations 5.5 Implications for literature 5.6 Implications for research 5.7 Implications for practice 5.8 Conclusion 154 155 157 160 163 165 168 169 References 171 Appendices 195 Appendix A - Study One Information Sheet Appendix B - Study One Consent Form Appendix C - Study One Interview Schedule Appendix D - Study One Debrief Sheet Appendix E - Study One University Ethics Committee Approval Appendix F - Information Sheet (Studies Two, Three and Four) Appendix G - Consent Form (Studies Two, Three and Four) Appendix H - Debrief Sheet (Studies Two, Three and Four) Appendix I - Adaptation to IBD Questionnaire (Study Two) Appendix J - Brief Illness Perceptions Questionnaire (Study Three) Appendix K - Inflammatory Bowel Disease Questionnaire (Study Three) Appendix L - Inflammatory Bowel Disease Questionnaire Stoma (Study Three) Appendix M - Study Four Interview Schedule Appendix N - University Ethics Committee Approval (Studies Two, Three and Four) 195 199 200 201 203 204 208 209 211 213 214 221 232 234 Index of tables Table 1. Participant demographic information 62 Table 2. Scale development and validation steps included and not included 86 Table 3. Factor loadings for 18 item adaptation questionnaire 95 Table 4. Means and standard deviations for 18 item adaptation questionnaire 96 Table 5. Correlations between A-IBD, Brief IPQ and IBDQ/IBDQ-S 100 Table 6. A-IBD and BIPQ predicting IBDQ scores 112 Table 7. A-IBD and BIPQ predicting IBDQ-S scores 113 Table 8. Participant demographic information 128 Table 9. Themes and subthemes of thematic analysis 131 1 Chapter One Literature Review 1.1 Definition of IBD Inflammatory Bowel Disease (IBD) is an umbrella term that encapsulates two very similar yet distinct idiopathic, chronic diseases of the gastrointestinal system: Ulcerative Colitis and Crohn's Disease. The validity of this grouping term is justified by the fact that both diseases are characterised by inflammation and ulceration of the gastrointestinal tract, frequent recurrences, and many gastrointestinal and systemic complications (Kirsner, 1995). The two diseases do however possess individual nuances. Ulcerative Colitis is a diffuse disorder of the colon which always affects the rectum and extends proximally to include variable degrees of the rest of the bowel. If only the rectum is diseased then this is often referred to as 'proctitis', which may be followed by 'distal colitis' involving the left or 'distal' side of the large intestine and finally 'total colitis' if the disease has spread throughout the large intestine. The disease never extends beyond the caecum (the intersection between the large and small intestine) and thus does not involve the small intestine. With Ulcerative Colitis only the innermost lining of the bowel wall becomes inflamed. In contrast, Crohn's disease can affect any part of the gastrointestinal tract. Although most commonly found in the ileocaecal region involving the terminal ileum (the most distal part of the small intestine) and the caecum, it is possible but rare for the disease to manifest itself solely in the anus, where it is referred to as 'perianal Crohn's', the mouth,known as 'oral Crohn's', or in the upper gut region - the oesophagus, stomach or duodenum, in which case it is referred to as 'gastroduodenal Crohn's'. Crohn's disease may involve several segments of intestinal inflammation simultaneously (known as 'skip legions') with regions of histologically healthy bowel in between (Jewell et al., 1992) and unlike Ulcerative Colitis, inflammation in Crohn's 2 disease is transmural, i.e. all layers of the bowel wall may be inflamed. Patients with Crohn's disease also frequently suffer from strictures of the bowel (also known as 'stenosis', a narrowing of the intestinal wall) or fistula formation (ulcers that extend completely through the intestinal wall creating abnormal connections between different body parts) into adjacent loops of the intestine (enteroenteric or enterocolic fistulas), the bladder (enterovesical or colovesical fistulas), the vagina (enterovaginal fistulas), the skin (enterocutaneous fistulas), often occurring following surgery along the incision line on the abdomen, or most commonly the anal area (perianal fistulas) (Crohn's and Colitis UK, 2013). While the areas of the gastrointestinal tract that the two diseases may inhabit differ, their symptoms share more similarities. The symptoms that IBD patients suffer from are commonly characterised by phases of exacerbation and intermittent remission (Jewell et al., 1992). During periods of disease relapse, symptoms that are commonly experienced by patients are phases of acute abdominal pain, spasms, nausea, fever, fatigue, diarrhoea and rectal bleeding (Cooper et al., 2010; Daniel, 2001). Even in times of remission, which are usually scarce, fatigue still largely affects patients' daily functioning (Czuber-Dochan et al., 2013).In conjunction with this, some may suffer from extraintestinal manifestations (EIMs) of the disease that may include maladies of the skin (most commonly 'erythema nodosum' - a condition where the layer of fat underneath the skin becomes inflamed causing red rounded, tender nodules to form, and 'pyoderma gangrenosum' - where pustules form on the skin and enlarge, coalesce and ulcerate) joints (arthritis and spondylitis) and eyes ('episcleritis' - mild pain and redness of the thin layer of tissue between the conjunctiva and connective tissue that forms the sclera of the eye, and 'uveitis' - inflammation of the middle layer of the eye called the uveal tract) (Vermeire & Rutgeerts, 2005). As well as these dermatologic, musculoskeletal and ocular EIMs, manifestations of the hepatopan-creatobiliary system such as 'primary sclerosing cholangitis - inflammation of the bile ducts inside and outside the liver 3 causing scarring (Navaneethan & Shen, 2010)as well as renal and pulmonary manifestations such as 'nephrolithiasis' - the formation of kidney stones (Levine & Burakoff, 2011). These EIMs can be classified into two major groups: the first including reactive manifestations often associated with intestinal inflammatory activity and thus mimicking a pathogenic mechanism common with intestinal disease (of which arthritis, pyoderma gangrenosum and uveitis are examples), while the second includes many autoimmune diseases that are not considered specific to IBD but are associated with a major susceptibility to autoimmune diseases as IBD is thought to be (including spondylitis and alopecia) (Danese et al., 2005).Some form of these EIMs are observed in as many as 25-40% of patients with IBD (Bernstein et al., 2001). Ulcerative Colitis and Crohn's disease, the latter originally being known as 'regional ileitis' and eventually 'regional enteritis', most likely began their rise to prominence as among the more challenging of human illnesses several centuries ago as isolated cases, their ambiguous nature reflected in the decades it took to identify the diseases as distinct diagnoses from more common gastrointestinal issues. The beginnings of Ulcerative Colitis may even date back as far back as to the days of Hippocrates (Hippocrates, 1886) who is said to have recognised different forms of diarrhoea but obviously lacked the technology by which to identify the underlying physiological issues and formulate diagnoses. The later epidemics of dysentery and bloody diarrhoea in Colonial America (Kirsner, 1990) were almost certainly of an infectious nature, the exact cause lost somewhere in the complex mixture of infectious colitides such as 'Clostridium difficile', 'Esherichia coli', 'Shigella', 'Salmonella', 'Campylobacter' and 'Colonic Tuberculosis', to name but a few. The plausibility of "nonspecific inflammatory bowel disease" was retrospectively suggested in an account of a 40 year old Irishman who in 1756 developed a persistent "bloody flux", but it wasn't until 1793 in British pathologist Matthew Baillie's Morbid Anatomy of Some of the Most Important 4 Parts of the Human Body that it was suggested that patients were dying from Ulcerative Colitis (Kirsner, 2001). The first "impact" description of "simple Ulcerative Colitis" was put forward by Samuel Wilks, a physician at Guy's Hospital in London, in 1859 (Wilks, 1859) who observed transmural ulcerative inflammation of the colon and terminal ileum upon performing an autopsy on a 42 year old woman who had died after several months of diarrhoea and fever. A century later this diagnosis would in fact be later identified as Crohn's disease (Fielding, 1985) and it wasn't until 16 years later in 1875 that Wilks described complete inflammation and a scattering of ulcers in the colon of a young woman who had died from severe bloody diarrhoea that "simple Ulcerative Colitis" was formally identified. The proper diagnosis of this disease became increasingly possible soon after with the introduction of the electric sigmoidoscope that enabled health care professionals to more readily distinguish it from illnesses such as 'infective dysentery', 'catarrhal Colitis', one of the milder forms of Ulcerative Colitis, or nervous diarrhoea (Baron, 2000). Indications of Crohn's disease meanwhile began to surface in the early 17th century when an autopsy of a boy who had died after persistent abdominal pain and diarrhoea and was described as having an 'ulcerated caecum' that was 'contracted and invaginated in the ileum'. Similar observations were later made by the likes of Fenwick (1889) who encountered "adherent loops of intestine with a communication between the caecum and adherent small intestine" along with a dilated and hypertrophied ileocaecal valve that "was contracted to the size of a swan's quill" in the autopsy of a 27 year old woman with a history of diarrhoea and weight loss. The descriptions of Crohn's then began to rise significantly in the early 20th century with case reports predominantly from Europe and North America documenting the occurrence of tumour-like granulomas (a collection of immune cells that cause inflammation) of the small intestine that were initially regarded as malignant and required surgical removal (Kirsner, 1988). The first clear description of the disease and the recognition that the granulomatous inflammation was 5 not tumour-related came from the work of Dalziel (1913) who described that one of his patients had experienced bouts of cramping abdominal pain and diarrhoea which progressed to intestinal obstruction and death with an autopsy that revealed entire chronic inflammation of the small intestine. This work actually preceded the seminal paper of Burrill Crohn, Ginsburg and Oppenheimer (1932) in which they described eight cases of regional ileitis from which the disease eventually derived its name. Confirmation that the disease was not localised solely to the ileum was later documented by Lockhart-Mummery and Mason (1960), yet the label of 'Crohn's disease' would remain. 1.2 Prevalence of IBD Although IBD initially rose as isolated cases in Great Britain and northern Europe, as well as more industrialised countries such as North America, the diseases have steadily increased numerically and geographically and today are recognised worldwide to now include reports from Asia, Africa and Latin America (Hanauer, 2006), suggesting it is now a global disease. This statement is backed up by Molodecky et al.'s (2012) time-trend analysis data which demonstrated that 75% of CD studies and 60% of UC studies included in a systematic review had an increasing incidence of statistical significance (p<0.05). Current epidemiological studies suggest that, in the West, the incidence and prevalence of IBD has increased in the past 50 years, up to 8-14 per 100,000 persons and 120-200 per 100,000 persons for UC, respectively, and 6-15 per 100,000 persons and 50-200 per 100,000 persons for CD, respectively (Cosnes et al., 2011; Meyer, 1984). It is now estimated that, in total, roughly 1.4 million people suffer from IBD in the United States and 2.2 million in Europe (Loftus, 2004). Specific examples of the increasing commonness of IBD have been illustrated by the likes of Munkholm et al. (1992) who found that the incidence of CD had increased six fold from 1962 to 1987 in Copenhagen, Denmark. These figures were later backed up in a prevalence study conducted by Vind et al. (2006) who found incidence rates of 8.6 per 6 100,000 for CD and 13.4 per 100,000, confirming the aforementioned current incidence estimations in the West. Indeed, Scandinavia appears to be a region in which IBD has become an increasing problem. Figures for CD from Stockholm, Sweden collated by Lapidus et al. (1997) had seemed to stabilise at 4.6 per 100,000 persons but, upon a recent review of these figures gathered during the period of time from 1990 until the turn of the millennium by Lapidus (2006), the statistics reflect those of the previously mentioned Danish studies, demonstrating that CD rates of incidence had markedly increased to a mean incidence rate of 8.3 per 100,000 persons, with 0.2% of the population being found to suffer from CD. Further to this, until recently there has been little data available on the epidemiology of IBD in Eastern Europe, but new studies from Hungary and Croatia are reporting steep increases in IBD incidence suggesting they are now comparable to Western European countries (Burisch, 2014; Lakatos et al., 2004). In conjunction with this, as mentioned the rest of the World alarmingly seems to be catching up to Europe too. Figures on the incidence and prevalence of IBD in Canterbury, New Zealand provided by Gearry et al. (2006) reflect the previously mentioned general Western statistics with UC incidence rates at 16.5 per 100,000 persons and CD rates of 7.6 per 100,000 persons, suggesting that IBD is at least as common in Canterbury as in Western regions where it was once suggested incidence and prevalence was low (Schlup et al., 1986; Eason et al., 1982). Meanwhile, age-adjusted prevalence figures from Lebanon report figures of 106.2 per 100,000 persons for UC and 53.1 per 100,000 persons for CD (Abdul-Baki et al., 2007), echoing similar prevalence rates to the West. Furthermore, although the epidemiologic profile within some countries is still developing, it is thought that even in these regions IBD is not as rare as previously thought, such as in Iran (Aghazadeh et al., 2005) where UC is of particular predominance, outnumbering CD cases at a rate of 9:1, and Korea where the incidence and prevalence rates of CD specifically have been thought to be increasing rapidly (Lee & Lee, 2014).Indeed, figures from Asia generally, but particularly 7 East Asia suggest an increasing incidence and prevalence of IBD over the past decade (Thia et al., 2008) with a twofold to threefold increase in incidence in several Asian countries (Ng, 2015). Even in regions where absolute incidence figures may not match those mentioned above, they are still noteworthy as indicators of IBD as an increasingly worldwide issue. The incidence of CD in Puerto Rico was recently investigated for the period 1996-2000, showing an increase from 0.49 to 1.96 per 100,000 persons, with a similar increase but double incidence for UC (Appleyard et al., 2004). Similarly, a report from Saudi Arabia from 19832002 showed an increasing median annual incidence for the period of 1993-2002 (1.66 per 100,000 persons) compared to 1983-1993 of more than 500% (Economou et al., 2009). Worryingly, but perhaps unsurprisingly, the escalating rates of paediatric IBD and in particular of CD have also been demonstrated in both developed and developing countries in a systematic review conducted by Benchimol et al. (2011). They found that of 28 studies that used statistical analyses to assess trends over time, 77.8% reported statistically significantly increased incidence of paediatric IBD, with 60% of these studies reporting a significantly increased incidence of CD and 20% reporting significantly increased incidence of UC. In England alone, recent figures from the Health and Social Care Information Centre stated that the number of teenagers with CD has jumped more than 300% in the last 10 years, from some 4937 16 to 29 year olds who were admitted for treatment between 2003 and 2004, rising to 19,405 in 2013 (Knapton, 2014). This suggests IBD is likely to become an increasing health threat to future generations, compounded by the indication that CD may be becoming more common than UC and with that coming the issues that arise with treating the more complicated, aggressive nature of CD. Besides the outlined international differences in both the incidence and prevalence of IBD, intranational differences also exist, further complicating healthcare professionals' understanding of and ultimately pursuit for a cure of the disease. For the most part, these 8 differences within countries tend to be associated with ethnicity. Sewell et al.'s (2010) study of a diverse group of IBD patients in an ethnically diverse healthcare system in San Francisco, USA, found that Asians and Hispanics were diagnosed at older ages (41.0 and 37.1 years, respectively) than whites (30.5 years) and blacks (31.7 years). Furthermore, CD was more common among blacks (50% of patients) than Asians (25.5% of patients) and a significantly higher proportion of whites had a family history of IBD (25.7%) than blacks (8.8%, p=.04) or Asians (5.9%, p=.003). These ethnic differences have been found to not be an artefact of ethnicity itself however, as studies have by the likes of Damas et al. (2013) examined the phenotypes of IBD in Hispanics in the USA and demonstrated differences in disease presentation between US-born and foreign-born Hispanics. Among the significant differences found were that foreign-born Hispanics were diagnosed at an older age than USborn Hispanics (45 vs 25 years of age) and manifested more UC (59.9% vs 41.0%).Similarly, and perhaps most well-established is the recognition that UC is three to five times more common in Jews than non-Jews living in Western countries (El-Tawil, 2009). Jews are likely particularly susceptible genetically to UC which will be elaborated on further in the following section on the supposed causes of IBD, but explaining research that has found that in Israel, Ashkenazi Jews have a higher incidence of UC than Sephardi Jews, but a lower incidence than Ashkenazi Jews in the USA or Northern Europe, complicates matters and suggests a complex genetic and environmental interplay which will again be discussed further. In terms of a gender bias in IBD, it is thought that women face a 20-30% greater risk of developing CD than men (Loftus et al., 2002), with recent figures from a North American study finding IBD was more prevalent in females than males (417 per 100,000 compared to 284 per 100,000) (Betteridge et al., 2013). However, this gender bias has not been supported worldwide and may be a characteristic of Western IBD as Ye et al. (2010) have found the opposite to be true with male-to-female ratios of 2.2:1, while Hu et al. (2014) reported a 9 male-to-female ratio of 1.8:1 in CD patients in their recent geographic mapping study of CD in an eastern province of China. Peak incidence for UC and CD occurs between the ages of 20 and 34 years (Gearry et al., 2006). IBD is thus typically perceived as a younger person's illness with an even more worrying incidence of IBD in childhood and adolescence which occurs in 25% of patients and is characteristically extensive and progresses quickly, impacting growth and development (Van Limbergen et al., 2008). Some studies have also found a bimodal distribution of IBD incidence, with another peak in the number of cases of IBD onset occurring at around 60 years of age (Ananthakrishnan & Binion, 2009). Finally, there is a relationship between IBD and a number of unfavourable socioeconomic variables, highlighted by Bernstein et al. (2001). Namely, patients with IBD are likely to be unemployed, with CD being more impactful in terms of the ability to maintain employment than UC. Similarly, and unsurprisingly, patients with IBD, and especially those who have undergone surgery, take more sick leave than their healthy counterparts (Marri & Buchman, 2005). Despite this, patients with IBD have a low rate of reporting themselves as disabled (1.3%). Fewer patients with IBD also achieved postsecondary education and among those married while living with IBD, approximately 10% of men and up to 20% of women were no longer married 5 years later. There is a slightly paradoxical relationship between socioeconomic status and IBD occurrence however, with some reporting that diagnoses of IBD are more frequent in those with a slightly higher socioeconomic status (Blanchard et al., 2001). 10 1.3 Causes of IBD Current theories on the cause of IBD are an amalgamation and interplay of environmental, infectious, genetic and immunologic factors (Fiocchi, 1998). This makes the identification of a cure an extremely difficult task due to the high probability that even if a single initiating factor is found for CD or UC, there are likely numerous complex interactions that may trigger and exacerbate the illness. In terms of understanding the genetic and immunologic factors that may underpin IBD, genome-wide association studies - studies that examine common genetic variants associated with a particular disease have identified approximately 100 loci (specific location of a gene, or significant DNA sequence, on a chromosome) that are significantly associated with IBD. These loci that have been implicated as factors in the pathogenesis of IBD include a diverse array of genes and pathophysiologic mechanisms including microbe recognition, lymphocyte (white blood cell) activation, cytokine (protein cells that aid cell to cell communication in immune responses and stimulate the movement of cells towards sites of inflammation) signalling, and intestinal epithelial defense (cells responsible for mediating intestinal homeostasis) (Cho & Brant, 2011). While technological advancements have revolutionised the understanding of complex genetics influencing diseases, the genetics and immunology of IBD are particularly complicated due to some susceptibility loci being specific to CD and UC while others are associated with both (Thompson & Lees, 2011). For example, an interleukin (a group of cytokines important in stimulating immune responses such as inflammation) known as IL-17 is strongly suggested to contribute to the development and progression of UC (Li et al. 2014), as well as immune function-related genes such as IGHG3 and IGLL2 (Lawrance et al., 2001). Meanwhile, for CD, defective processing of intracellular bacteria and interactions with intestinal flora which regulate inflammatory response by stimulating the immune system has been suggested as key to its pathogenesis, 11 with the NOD2 and ATG16L1 genes (Cho, 2008), along with elevated Interleukin-12 levels (Mannon et al., 2004), are implicated as culprits contributing to this breakdown. One reason why understanding IBD poses such a difficult task however is that certain gene regions such as STAT3 and NKX2-3 are associated with both CD and UC indicating common but also unique mechanisms for the genesis of IBD. This is compounded by the fact that many immune-mediated diseases, as IBD is largely suggested to be and an argument that is supported by the aforementioned genes being responsible for regulating immune function, indicate a genetic overlap, such as with type 1 and type 2 diabetes mellitus (Lees et al., 2011). Similarly, the IBD diagnoses of patients from different backgrounds have been found to not necessarily be related to commonly implicated genes. For example, Ouyang et al. (2005) found a remarkable difference in the CD diagnoses of patients from the Asian Pacific region, stating that there was an absence of any association with Asian CD with the aforementioned NOD2 gene as has been repeatedly observed in white and Jewish patients. In conjunction with this, Halfvarson et al.'s (2003) study on monozygotic twins found that the concordance level of IBD was only 50% for CD and 19% for UC, reiterating further the need to understand more than genetics of IBD in order to explain how it is borne. Even as cutting edge research has revealed that CD and UC are also associated with the absence of a gene known as 'uhrf1', which is said to be responsible for preventing TNF (tumour necrosis factor) molecules from signalling pro-inflammatory immune signals into the intestinal barrier, it is still not clear what triggers increased TNF levels in the guts of patients with IBD or how this event leads to disease onset (Duke University, 2015). Others argue that the pathogenesis of IBD can be attributed to infectious agents. In a population based study from two Danish counties comparing those exposed to salmonella and Campylobacter gastroenteritis with unexposed persons, the hazard ratio of the onset of IBD for those exposed compared to unexposed was 2.9 (95% CI 2.2-3.9) for the whole 7.5 year 12 observation period and 1.9 (95% CI 1.4-2.6) for the first year (Gradel et al., 2009). Other infectious exposures have been argued as being beneficial in protecting from IBD. For example, Luther et al. (2010) in their systematic literature review suggested that there is a protective effect of Helicobacter pylori infection against the development of IBD, demonstrating that in the 23 studies that were included in their review, 27.1% of IBD patients had evidence of H. pylori infection compared to 40.9% of control group patients. More generally, it is also hypothesised that "cleaner" environments with lower rates of communicable diseases are associated with higher rates of IBD onset, with the opposite also being true. As such, protective factors against IBD that have been suggested in accordance with this hypothesis are lower usage of antibiotics, increased number of pets and livestock, larger family sizes, and increased exposure to pathogens such as the aforementioned H. pylori (Ponder & Long, 2013).Indeed, this theory has been attributed to the increasing incidence of IBD, as well as allergic reactions such as gluten intolerance, that are thought to be linked to the worldwide adoption of a contemporary westernised lifestyle (Wills-Karp et al., 2001). Following on from this, other behavioural and lifestyle factors such as smoking, diet and stress are also thought to have an impact on the risk of IBD genesis. The issue of smoking and its link to IBD pathogenesis has been extensively researched is now the most well understood of the three contributors listed. However, in line with the ambiguity of IBD, smoking has been found to have a clear opposite effect on CD and UC with it proving to be a risk factor for CD, increasing the frequency of disease relapse and need for surgery while being a protective factor in UC, with cessation of smoking increasing the risk of developing UC ((Rubin & Hanauer, 2000). Once again the particular mechanisms by which each form of IBD occurs, namely CD and UC, is shrouded in uncertainty (Thomas et al., 1998). Dietary factors meanwhile provide a far less clear picture of their link to IBD onset, but given the importance of food in everyday life along with the increasing vigilance of artificial 13 components used in the manufacture of food it undoubtedly warrants investigation. Furthermore, given the fact that IBD is a gastrointestinal disorder, it is reasonably safe to presume that dietary factors are likely to be at least influential. However, as stated, this logic is as of yet not definitively proven, mostly because establishing a cause and effect relationship between dietary factors and IBD is extremely complex and defining the true composition of a diet is problematic (Danese et al., 2004). Studies that have attempted to indicate diet and IBD associations include Ananthakrishnan et al.'s (2014) study which suggested that high intake of trans-unsaturated fats may be linked with an increased risk of UC, while Andersen et al. (2012) suggested a diet high in protein, particularly animal protein, may be associated with increased risk of IBD and subsequent relapses. Yet advanced research into dietary hypotheses, such as the 'FODMAP' hypothesis, may be bringing us closer to the answer on whether certain types of sugars in particular, implicated in research as far back as Sonnenberg (1988) are indeed a culprit in IBD, or at least in CD. The FODMAP (Fermentable Oligo-, Di and Mono-saccharides, and Polyols) hypothesis states that CD susceptibility may be associated with an excess in the delivery of highly fermentable but poorly absorbed short-chain carbohydrates and polyols (sugar alcohols, reduced-calorie carbohydrates that provide the taste and texture of sugar) to the small intestine and lumen of the colon which rapidly ferment in the bowels and leads to increased intestinal permeability, which as discussed previously, predisposes one to CD onset. It is argued that this hypothesis is supported by the increasing intake of FODMAPs in Western societies (Gibson & Shepherd, 2005). Or perhaps the escalating issue of IBD is due to the rising use of emulsifying agents in our foods. Emulsifiers, a common component of processed foods, are detergent-like molecules that may cause IBD by damaging the mucosal barrier and thus allowing gut bacteria to interact with epithelial cells that line the intestinal walls. Chassaing et al. (2015) recently found that relatively low concentrations of two commonly used emulsifiers induced 14 low grade inflammation and promoted robust colitis in mice predisposed to this disorder. With increasing globalisation of the Western diet, it is arguable that these emulsifiers may underlie the ever more global issue of IBD. That being said, obviously correlation does not necessarily equal causation, and thus such hypotheses remain just that for the time being. Finally, and perhaps most controversially is the association of oral contraceptives with IBD pathogenesis, and CD in particular which has garnered much attention in the media as of late. The reports link back to certain studies such as that of Khalili et al. (2013) who found that compared with never users of oral contraceptives, the adjusted Hazard Ratio for patients with CD was 2.82 among current users and 1.39 amongst past users. The direct association between oral contraceptives and UC was less certain as women who had previously or currently smoked were found to be at a higher risk of UC, but there was no significant association among women who never smoked. This starkly contrasts Khalili et al.'s (2012) previous prospective cohort study findings that women obtaining or who had previously obtained postmenopausal hormone therapy appeared to show an increased risk of UC (adjusted Hazard Ratios of 1.71 and 1.65, respectively), while there was no observed association between current use of hormones and risk of CD pathogenesis. Most controversially of all explanatory factors for the pathogenesis of IBD is the focus of stress as a trigger. A belief in the relevance of psychological factors dates as far back as the 1930s when gastroenterologists and psychiatrists considered IBD a psychosomatic disease and that emotional life events and experiences were linked to intestinal symptoms (Keefer et al., 2008). Methodological issues in assessing the causative nature of stress in IBD has since largely quashed it as a plausible explanation for the manifestation of the disease, yet, while purely anecdotal, almost 75% of patients suffering from IBD believe that stress or their own personality is a major contributor to the development of their illness (Sajadinejad et al., 2012).What is beyond doubt however is the role of stress in worsening already existing 15 IBD and the cycle of stress, symptom exacerbation, and subsequent further stress that many patients find themselves in as a consequence of attempting to manage life with IBD. 1.4 Consequences of IBD Living with IBD has both physiological and psychological consequences for the patient. These will now be discussed. 1.4.1 Physiological consequences In terms of mortality rates, IBD patient survival is similar to the general population. This fact appears to be true regardless of the time period during which patients are diagnosed as a recent prevalence cohort study of IBD patients in Australia diagnosed after 1970 conducted by Selinger et al. (2013) found that there was no difference in survival between patients diagnosed earlier (1970-1979) or later (1980-1992). Yet while CD or UC themselves are rarely directly implicated in the mortality of patients with IBD, Selinger et al. also found that these patients were more susceptible to fatal cholangiocarcinomas (bile duct cancer) (p<.001) and fatal colorectal cancers particularly in UC (p=.047). Indeed, colorectal cancer (CRC) is generally found to be markedly more of an issue in patients with ongoing UC and CD and particularly in those who present with certain risk factors such as greater anatomic extent of UC, a more severe degree of inflammation, and have the presence of primary sclerosing cholangitis and family history of colorectal cancer. Meanwhile, CD patients suffering from disease activity in the small intestine also at an increased risk of small bowel adenocarcinoma (cancer of the mucus-secreting glands in the epithelial tissue) (Triantafillidis et al., 2009).Besides being more susceptible to CRC, in a comparison of CRC patients with and without UC, UC-CRC patients with stage III colorectal cancer had a poorer survival rate than the sporadic CRC patients (43.3% versus 57.4%, respectively, p=.0320) (Watanabe et al., 2011). The fact that the risk of CRC increases 8 to 10 years after a diagnosis of UC 16 (Yashiro, 2015) is a strong suggestion that the increased risk of cancer in IBD is a consequence of the disease rather than an inherited phenomenon, most likely due to factors such as oxidative stress and apoptosis (cell death) resulting from prolonged inflammation (Breynaert et al., 2008). Despite the largely non-life threatening outlook of being diagnosed with IBD, patients instead face the prospect of multiple acute or ongoing threats to their health as a consequence of their diagnosis. First and foremost is the issue of pain in IBD and its management which is a frequent issue amongst the IBD population, in both an acute and chronic sense. Medical professionals' most obvious task is that of pain amelioration and yet it is often regarded as the most challenging aspect of their role in providing care for the patient (Giordano & Schatman, 2008). This difficulty is augmented when dealing with an illness such as IBD for which it is often a complex matter to identify patient's pain as a result of the potential for pain arising due to a variety of underlying issues: active inflammation, secondary complications, as well as functional pain that has no structural or biochemical explanation (Docherty et al., 2011). Even when patients achieve a period of remission from their IBD, although the exact definition of this status can vary, Simren et al. (2002) found that in a study of 43 patients in remission from UC and 40 patients in remission from CD, 33% and 57%, respectively, were experiencing symptoms of Irritable Bowel Syndrome (IBS, a syndrome that is characterised by pain and symptoms akin to IBD, albeit less severe, but not accompanied by inflammation or other tissue-related changes). Crucially, those experiencing these IBS symptoms displayed decreased levels of wellbeing than those without, and so it becomes apparent that gastroenterologists' and accompanying healthcare practitioners' task of providing improved quality of life for their patients may not be accomplished even when they may manage to help control the IBD itself. 17 In conjunction and as previously highlighted, patients with IBD are prone to suffering inflammatory co-morbidities that are also often associated with pain such as fibromyalgia which has been documented by Buskila et al. (1999) to be found in around 30% of patients with IBD. More general extraintestinal manifestations associated with IBD were found to appear at least once in as many as 46.6% of patients in a prospective study of 295CD and 271 UC Spanish sufferers, with joint manifestations the most common type (Mendoza et al., 2005). Indeed, IBD-related arthropathy is the most common extraintestinal manifestation reported in the IBD population and can have a significant impact on morbidity and quality of life. More specifically, those suffering from arthropathy are considered to be afflicted with an axial variant which may include inflammatory back pain, sacroiliitis (inflammation of the joint formed by the union of the sacrum and the ilium), or ankylosing spondylitis, which is less likely to correlate with gastrointestinal symptoms; a peripheral variant which is generally non-erosive and may correlate with one's disease activity; or a combination of both types. As such, treatment of one's IBD symptoms may not always have the same beneficial effects for controlling one's arthritic symptoms (Arvikar & Fisher, 2011). Treatment of this arthritis can be achieved alongside IBD disease activity through the use of biologics that temper the body's inflammatory response, however short term use of non-steroidal anti-inflammatory drugs (NSAIDs) (De Vos, 2009), yet these can in turn irritate the gut lining and trigger one's IBD symptoms, further highlighting the difficulty in managing IBD in that it rarely results in intestinal symptoms in isolation. Despite facing the prospect of extraintestinal manifestations of their IBD and the possibility of unwanted side effects of their treatment, patients' most pressing concerns in this facet of life with IBD arise from the side effects of their main treatment regimens. While major advancements have been made in developing medications for IBD which have, for the most part, been largely successful in circumventing suffering that is a direct result of active 18 disease, largely due to the rise of biologic agents which target specific inflammatory agents that are thought to be key in the over active immune system of patients with IBD (Triantafillidis et al., 2011), their side effects can be troublesome at best, and life threatening at worst. Infections and septicaemia are the most common threats associated with this treatment, and in fact with treatments lower down the 'pyramid' of IBD treatment such as corticosteroids such as prednisone/prednisolone and more general immunomodulators (as opposed to target specific modulators in the case of the biologic drugs) such as Azathioprine or Methotrexate. The issue here is that biologics augment these risks as they are often combined with immunomodulators for greater efficacy by preventing antibody formation to the drug (D'Haens, 2007). Of particular concern are the opportunistic infections that patients being treated with biologics and immunomodulators may encounter, such as listeriosis (caused by eating food contaminated by listeria bacteria), nocardiosis (most commonly an infection of the lungs, but also sometimes of the skin, brain, or other organs) and invasive aspergillosis (a fungal mould that usually affects the respiratory system but can spread to any region of the body) to name but a few (Blonski & Lichtenstein, 2006).The risks of such infections were investigated by Toruner et al. (2008) who found that Azathioprine/6Mercaptopurine (known as 6-MP, another 'thiopurine' immunomodulator) was associated with an OR of 3.1 for developing opportunistic infections, Infliximab (a brand name for one of the more popular biologics) with an OR of 4.4, but the combination of the two producing a potential OR of 14.5, depicting one clear drawback of a combination therapy of immunomodulators and biologics. Other possible adverse events include neurological disorders caused by demyelinisation in patients treated with biologics, including reports of multifocal motor neuropathy (a rare condition in which multiple motor nerves are attacked by the body's immune system causing muscles to gradually weaken) (Rutgeerts et al., 2005) and optic neuritis, the latter of which dissipated entirely after discontinuation of the treatment 19 (Hommes et al., 2005). Other similarly drastic concerns include the potential for the reactivation of latent tuberculosis and hepatitis B, congestive heart failure (Parekh & Kaur, 2014) as well as liver toxicity (Carvalheiro et al., 2013). Most worrying of all potential adverse outcomes from immunomodulating therapy however is the risk of cancer development, particularly with regards to hematologic malignancies such as lymphomas. Thiopurines alone have been found to be associated with increased risk of non-Hodgkin lymphoma in particular, the addition of anti-TNFs also significantly increases the risk of a rare hepatosplenic T-cell lymphoma, particularly in young males with CD (Biancone et al., 2015). While the absolute rates of these malignancies remain low((Siegel et al. (2006) observed 201 cases of lymphoma in 100,000 patients at 1 year while receiving Infliximab)), help reduce steroid dependency (Schintzler et al., 2009) and have a major impact on key disease parameters such as fewer hospitalisations and surgical procedures (Lichtenstein et al., 2005), thus suggesting the potential benefits of these treatments outweigh the potential risks, the impact of these aforementioned potential side effects on both a physical and psychological front for the patient must not be downplayed. The possibility for serious consequences are undoubtedly a factor to consider in terms of safeguarding both the physical and psychological wellbeing of patients. This is particularly true given the non-curable nature of IBD and the fact that these drug treatments are often prescribed indefinitely even though the ideal and safest duration of treatment as well as the identification of which patients may stop therapy is yet to be fully understood (Clarke & Regueiro, 2012), and withdrawal can substantially increase the risk of relapse (Gomollon & Lopez, 2008). What's more, for those who do withdraw, relapse, and then wish to commence biologic treatment once again, the risk of immunization or hypersensitivity reactions should be taken into account (Magro & Eliakim, 2014). Finally, the need for precise management of these drugs through the implementation of administration schedules over episodic/on demand treatment has been 20 proven by Baert et al. (2003) who found that the latter approach led to antibody formation in >70% of patients treated with Infliximab, which means these patients may often have to resort to other drugs in emergencies such as the above mentioned corticosteroids which bring with them a whole host of other side effects including acne, severe mood changes, visual changes due to steroid-induced hyperglycaemia, early cataract formation and potentially even aspectic joint necrosis (death of bone matter) (Botoman & Bonner, 1998). While such valuable discoveries have been made and continue to be made over the safety of these medications, patients still face great uncertainty as to whether their bodies will tolerate these strong drugs and, if so, for how long. Yet even when drug treatment is successful, fatigue then becomes the most troublesome symptom for many patients in remission, occuring in more than 40% of cases (Minderhoud et al., 2003). The mechanisms by which this fatigue occurs are not well understood (Czuber-Dochan et al., 2013) and this is likely due to its prevalence during periods of not only quiescent disease activity, but no remaining residual anaemia or iron deficiency being evident (Bager et al., 2012). Such fatigue has been found to have a significant association with depression (Grimstad et al., 2015), disability (Cohen et al., 2014) and health-related quality of life (Romberg-Camps et al., 2010). This issue is compounded further by an obvious gap in healthcare professionals' knowledge and understanding of the complexity of IBD fatigue and the gravity of its impact on patients' lives (Czuber-Dochan et al., 2014). Furthermore, however successful modern medicines have proved to be in reducing the need for surgical intervention, this is another reality that patients often have to confront as it is still highly likely that patients will have to undergo numerous and sometimes drastic, emergency surgeries (particularly in the case of UC patients who opt to undergo a colectomy or proctocolectomy in order to essentially 'rid' themselves of the disease as UC is localised to 21 the large intestine and rectum). In general, approximately 70% of people with CD will need some form of surgical intervention within 10 years of diagnosis (Peyrin-Biroulet et al., 2010) and around 20% of patients with UC will require a colectomy following an acute attack (Artari et al., 2008). Even in this era of biologics, approximately one in five patients treated with these powerful drugs require intestinal resection (removal of a portion of the bowels with subsequent reattachment of the remaining bowel) after 2-5 years of this treatment (Peyrin-Biroulet & Lemann, 2011). The financial burden of IBD that such surgeries and drug treatments place on both governments and patients themselves who suffer from greater rates of unemployment (around 10%) and take more sick leave (3-6 weeks per year) and experience around twice as much permanent work disability as a result of their illness, must also be taken into account when assessing the challenges IBD poses. In Europe alone it is estimated that there is a total yearly direct healthcare cost of 4.6-5.6 billion Euros (Burisch et al., 2013). Recent figures suggest that for CD alone, when European and USA data is combined, annual total costs reach almost 30 billion Euros with more than half of that sum due to indirect costs (Floyd et al., 2015). 1.4.2 Psychological consequences These physiological and financial obstacles in turn contribute to and are intertwined with the psychological challenges of living with the chronicity of IBD, which may have negative social and behavioural implications. For instance, with particular respect to the fact that surgery is commonplace in IBD, patients often have to live with significant abdominal scarring and possibly stomas and ostomy bags that may even be required or opted for on a permanent basis. Saha et al. (2015) conducted a study of adults aged 18 and above who had been diagnosed within the past six months and followed them up over a two-year period to determine whether body image dissatisfaction (BID) changed over time in patients with IBD, finding that in 274 patients studies BID was found to be stable over time for both men and 22 women and across IBD subtypes despite overall improvements in disease activity. BID is particularly linked to steroidal treatment,due to the possibility of aforementioned facial swelling and acne, and has a number of repercussions for the patient including low levels of general and IBD-specific quality of life, self-esteem and sexual satisfaction. Consequently, BID concerns in patients with BID are multipronged in that they are not only physical but psychosocial too, affecting crucial aspects of life including work and social activities (McDermott et al., 2015), but also more unspoken facets of life such as patients' intimate relationships. This is particularly true as BID has been found to be more prevalent amongst women with Muller et al. (2010) reporting that 74.8% of versus 51.4% of males complained of an impaired body image. The knock on effect of this being that a greater proportion of operated women also reported decreased frequency of sexual activity versus males as well as decreased libido. BID stemming from stomas play arguably the largest part in deterring these patients from engaging in sexual activity since disguising the stoma bag and worrying about it leaking interfere with one's ability to focus and ultimately make it a pleasurable experience for both themselves and their partner. Adjusting to sex can be troublesome at the best of times in the case of a patient with a long established partner, but can prove even more problematic should the patient be single and seeking a partner who they will then need to disclose the matter to, resulting in self-consciousness, and social unease (Manderson, 2005). This issue may then be compounded by the burden of incontinence that some patients must deal with, as well as more immediate boundaries to social interaction such as crippling abdominal pain, which may consequently lead to complete social isolation as opposed to mere unease (Karwowski et al., 2009). Unsurprisingly, this concoction of negative factors associated with life with IBD consequently contributes to raised levels of psychological comorbidities in this population. Walker et al. (2008) demonstrate this issue in their population-based study of the prevalence 23 of lifetime mood disorders in 351 patients with IBD and 779 region-matched controls. Their comparison suggested higher rates of panic, generalized anxiety, and obsessive-compulsive disorders and major depression in the IBD sample than in national samples from the United States and New Zealand. Most persistent of these issues are anxiety and depression. Recent findings by the likes of Tribbick et al. (2015) have found that almost 20% of patients have at least one anxiety condition, while just over 10% have a depressive disorder present, while figures reported by Bannaga and Selinger (2015) suggest abnormal anxiety levels are found in up to 40% of patients with IBD, and even up to 80% for anxiety and 60% for depression during a relapse phase (Addolorato et al., 1997). Even in remission rates of anxiety and depression have been found to be as high as 29-35% (Mittermaier et al., 2004). These psychological disturbances in turn are associated with negative quality of life and health outcomes such as predicting even lower sexual function (Timmer et al., 2007), increased disease activity (Graff et al., 2009), an increased risk of surgery in CD by up to almost 30% (Ananthakrishnan et al., 2013) as well as enhancing patients' perceived barriers to medication adherence (Gray et al., 2012), further perpetuating this deleterious cycle. The amalgamation of these biopsychosocial concerns result in decidedly low healthrelated quality of life (HRQoL) scores amongst the IBD population. Floyd et al.'s (2015) systematic review of 61 publications concluded that CD patients in the US and Europe experienced consistently and statistically significantly lower HRQoL compared with normal populations due to physical, emotional and social effects. Similarly, Bernklev et al. (2005) found that in a population-based cohort of 348 Norwegian patients with IBD as compared to 2323 Norwegian healthy controls, six of eight and seven of eight dimensions of the SF-36 were significantly lower in patients with UC and CD, respectively. Meanwhile, Janke et al.'s (2004) study on 415 IBD outpatients reminds us that this disease significantly impacts not only health-related quality of life, but general life satisfaction too in both CD and UC. These 24 results are, as is often the case, linked to disease severity, yet there is sufficient evidence to suggest that quality of life is also impacted during periods of remission. For example, Lix et al. (2008) found that changes in IBD QoL were influenced by one's longitudinal profile of disease activity, namely those who experienced fluctuating disease activity showed improvement over time versus those with consistent activity, but this profile had only modest effects on their psychological functioning with regards to aspects such as perceived stress, health anxiety and pain anxiety. Indeed, the importance of not assuming patient satisfaction purely based on disease activity was suggested as far back as Drossman et al. (1989) who found that patients' functional status and disease concerns correlated better with scores of wellbeing and healthcare utilisation than physicians' rating of their disease activity. Similarly, family members have been found to underestimate patients' self-reported HRQoL (Guyatt et al., 1989). What these results together suggest therefore is twofold in that, firstly, IBD has an impact even when patients are not necessarily experiencing active disease symptoms, and that quality of life as it is perceived by the patient themselves as well as absolute health should be of importance in the care of patients with IBD by families and medical health professionals. Clinical remission therefore is not always the answer in IBD, particularly when one also takes into account the persistent problem of fatigue highlighted earlier and the possibility for anxiety and depression to carry over from periods of disease activity to periods of inactivity and the subsequent effects these things have on functional and psychological status. IBD is thus clearly a disease that is all-encompassing for patients, challenging them on a multitude of levels and is a hindrance to quality of life at all degrees of disease severity and thus adaptation to it behaviourally, socially, psychologically and emotionally is logically the key to thriving with the illness, especially when one takes into account its chronic nature. Sajadinejad et al.’s (2012) review article on the psychological issues on IBD refers to the need for appropriate coping strategies and good adaptation in order to deal with the 25 symptoms of IBD and its psychological impact. However, whilst Sajadinejad et al. (2012) advocate the need for patients with IBD to be adapted to their illness, little is known concerning what good adaptation means to patients with IBD or how it can be measured. 1.5 IBD as a chronic condition IBD can pose multiple problems for patients including the symptoms of the disease itself, the likelihood of other extraintestinal autoimmune disorders, the high probability of surgery being required and its consequences all impacting the experience of living with IBD. Further, these physiological aspects of the disease are then often associated with psychosocial impacts which combine to significantly influence the quality of life of those individuals suffering from IBD. Consequently IBD, much like a multitude of other chronic illnesses, is very often an all-encompassing disease that challenges many patients not only on a daily basis but also on a number of levels. As a result, this means that in lieu of a cure much of the chronic illness literature is centred around the key psychological constructs of processes, namely coping and sense making, and outcomes, which for the purpose of this thesis will revolve around quality of life (QoL). In other words, since medical intervention has yet to find a cure for most chronic conditions, the psychology of chronic conditions has mostly addressed how patients make sense of their illness and what coping strategies best predict patients' level of QoL. It should be noted, however, that although coping and sense making will be discussed in this introduction as distinct constructs it is recognised that they are not mutually exclusive and that notions of coping are often embedded within models of sense making and vice versa. Furthermore, whilst this thesis will discuss QoL as an outcome of coping and sense making it is also recognised that the relationship between coping, sense making and QoL is iterative and dynamic. These different key aspects of process (coping and sense making) and outcome (QoL) will now be discussed. 26 1.6 The process of having a chronic condition The process of having a chronic condition has mostly been studied in terms of coping and sense making. 1.6.1 Coping Coping is generally viewed as a collection of purposeful, volitional efforts that are directed at the regulation of aspects of the self and the environment under stress (Compas et al., 2012). For example, Skinner and Wellborn (1994) defined coping as 'action regulation under stress', while Eisenberg et al. (1997) interpreted coping as involving 'regulatory processes in a subset of contexts - those involving stress'. More broadly though, and most reflective of the implicit link between coping and its regulation of one's psychological and physiological processes is Compas et al.'s (2001) definition whereby coping is conceptualised as 'conscious, volitional efforts to regulate emotion, cognition, behavior, physiology, and the environment in response to stressful events or circumstances'. The main challenge in conceptualising coping comes though when we attempt to specify the structure of coping responses since coping is not a specific behavior and rather a broad, umbrella construct that envelopes a wide range of behaviours and cognitions that one uses to try and overcome a stressful event. For example, lower order categories of coping may consist of strategies such as problem solving, rumination and venting, while these lower order categories can then be further categorised into higher order categories or 'families of coping' such as approach or emotion-focused coping (Skinner et al., 2003). Indeed, Lazarus and Folkman's (1984) paradigm of coping with stress differentiates between emotion-focused and problem-focused which has proved particularly useful for the chronic illness literature. Here it is argued that stress, in this context of the current thesis being produced as a product of chronic illness, elicits an appraisal of the situation at hand that involves, firstly, a primary appraisal of the 27 situation to assess whether the threat is potentially harmful, threatening, blocking of a goal, creating a void or presenting a challenge. If the stress is indeed deemed to be threatening on any of these levels then the individual begins a secondary appraisal process whereby one's resources for coping are examined. At this point, the individual decides whether to employ problem-focused coping efforts or emotion-focused coping efforts, with the former directed at controlling or changing the sources of the stress such as by learning new skills, removing barriers or generating alternative solutions, and the latter revolving around attempting to manage the emotional responses to the stressor by utilising, for example, wishful thinking, seeking emotional support or conducting social comparisons (Snyder, 1999). Neither approach is argued to be inherently and universally beneficial or detrimental however, rather it is argued that the individual is required to utilise the most appropriate approach given the particular stressors associated with a situation (Taylor & Stanton, 2008). Evidence of this phenomenon is abundant within the chronic illness literature, with the likes of Graue et al. (2004), for example, finding that poor metabolic control and a lower degree of diabetesrelated quality of life are associated with greater use of emotion-focused coping in those with type 1 diabetes, while Affleck et al. (1999) found that patients with osteoarthritis experienced improved pain levels following a day with more emotion-focused coping. Lazarus and Folkman's dichotomous paradigm of problem and emotion-focused coping which was later operationalised by their Ways of Coping checklist (Folkman & Lazarus, 1988), was subsequently elaborated on by Carver et al. (1989) in their COPE inventory. The COPE inventory consists of 15 scales and was developed with a view to further conceptualise coping as a broader process than merely revolving around the two dimensions of problem and emotion-focused styles (Litman, 2006). The 15 scales include five scales on problem-focused coping such as 'planning' and 'instrumental social support' which involves seeking advice from others, along with a further five scales on emotion- 28 focused coping such as 'positive reinterpretation', 'turning to religion' and 'denial', but is supplemented by "less useful" coping scales such as 'behavioural disengagement' and 'mental disengagement' as well as 'recently developed' coping scales such as 'substance use' and 'humour'. Moos and Schaefer (1984) meanwhile applied 'crisis theory' to their theory on the process of coping with chronic illness. Crisis theory, stemming from Erikson's (1963) early work on psychosocial crises that arise at different stages of human development, was used by Moos and Schaefer to demonstrate how illness is a crisis and represents a turning point in the life of an individual diagnosed with a chronic illness. They put forward the notion that physical illness causes a number of changes than can be considered as a crisis, be they a change in identity from breadwinner to a person with an illness, a change in role from independent adult to passive dependant or changes in one's future from one involving children, a career or travel to one shrouded by uncertainty. This can be compounded by illness-specific factors such as the unpredictability of the illness, unclear information regarding the illness, ambiguousness about the causality and potential outcome of the illness, as well as limited experience in dealing with illness in general (Ogden, 2004), the majority of which are true for those living with IBD and have indeed been highlighted in the previous section on the consequences of life with IBD. Once confronted with these crises, Moos and Schaefer argued that a three step process occurs by which individuals first cognitively appraise the disequilibrium that is created by the illness crisis, appraising the seriousness and significance of their illness. Following this, it is theorised that seven adaptive tasks are carried out which can be separated into, firstly, the illness related tasks of dealing with pain and other symptoms, dealing with the hospital environment and treatment procedures, and developing and maintaining relationships with health professionals. Meanwhile, general tasks involve preserving emotional balance, preserving self-image, competence and mastery, 29 sustaining relationships with family and friends, and preparing for an uncertain future. Following this, Moos and Schaefer described how a number of coping skills can be utilised to deal with the crisis of illness, such as problem-focused coping which they argued involves seeking information and support, taking problem-solving action such as learning how to administer insulin injections for oneself and identifying alternative rewards such as constructing goals that can provide short-term satisfaction. This is supplemented by emotionfocused coping which in this context involves efforts to maintain hope when dealing with a stressful situation, emotional discharge aimed at venting feelings of anger or despair, as well as resigned acceptance which requires one to come to terms with the inevitable outcome of an illness. Finally, appraisal-focused coping is employed in order to enable oneself to carry out a logical analysis of the event and mentally prepare to deal with the illness, cognitively redefine the situation into one that is positive and acceptable, as well as utilising cognitive avoidance and denial to minimise the seriousness of the illness. In summation, Moos and Schaefer's theory on coping with physical illness builds on Lazarus and Folkman's dichotomous theory of coping as involving either merely problem or emotion focused coping, including not only three varieties of coping style, but also cognitive appraisal and adaptive tasks to overcome the crisis of chronic illness. Interwoven within the coping literature, particularly evidenced by Moos and Schaefer's notions of cognitive appraisal and the general adaptive task of preserving selfimage, competence and mastery is the process of sense making in the face of an illness event. While the processes of coping and sense making are arguably overlapping, for the purpose of this thesis they will be addressed separately. 30 1.6.2 Criticisms of the coping literature While coping styles have garnered a lot of attention in relation to patient outcomes in the chronic illness literature, the habit of associating a particular coping method with a broader coping style has proven to occasionally be misleading in terms of determining what coping style is most predictive of positive outcomes. For example, emotion-focused coping as mentioned previously has been interchangeably termed as avoidance-focused coping and this has repeatedly been found to be linked to negative outcomes like depression in MS (Arnett et al., 2010), emotional distress in cancer patients (Dunkel-Schetter et al., 1992), poor psychosocial adjustment and depression in those with persistent low back pain (Bombardier et al., 1990). However, when one views avoidant coping in a more broad, neutral manner, to include, for example, appraising a situation in a way that focuses on potential positives and avoids the negatives, this may engender a greater sense of patient control which may facilitate adjustment rather than maladjustment (as advocated in the CAT principle of 'positive illusions' which will be explored further below). Support for this can be found in the literature on benefit finding in chronic illness which has shown that this coping method is predictive of positive outcomes in the form of less distress and perceived growth (Helgeson et al., 2006). As such, nomenclature such as problem-focused and emotion-focused coping (as well as its alternative labels of approach and avoidance-based coping) runs the risk of masking important differences within categories. For example, typically, mental disengagement which involves an effort to deliberately distance oneself from thinking about a problem, and escape-avoidance, which is more of an escapist flight that can include behaviours such as excessive substance use, are both avoidant forms of coping that are usually grouped under the 'emotion-focused coping' umbrella term. However, mental disengagement is often adaptive when one does not possess control of a situation such as the results of an exam (Folkman & Lazarus, 1985), whereas escape-avoidance is usually 31 maladaptive with regards to outcomes. (Folkman & Moskowitz, 2004). Indeed, reflecting on Moos and Schaefer's (1984) COPE inventory, neither mental disengagement nor substance use are referred to as emotion-focused coping strategies, rather being labelled as merely "less useful" and 'recently developed', exhibiting the issue in this area of literature of categorising individual coping strategies under broader coping style labels. This issue was aptly summarised by Skinner et al. (2003) whereby the literature has attempted to categorise lower order "instances" of coping into mutually exclusive, conceptually clear higher order "families" of coping which reflect the coping styles discussed (problem-focused, emotionfocused, approach-based, etc.). In this paper, Skinner et al. conclude that while the coping literature has produced an exhaustive, broad list of coping instances, it is when these instances have been grouped into higher order categories such as problem vs emotion-focused that are not good categories because any given way of coping is likely to serve many functions. Yet commonly used families of coping such as problem or emotion-focused coping are not exhaustive with respect to lower order categories of coping, with some important instances of coping falling outside both of these broad higher order categories. For example, social support seeking is commonly employed by individuals in a health crisis and yet appears to be focused neither on the problem nor on emotion but instead on other people, thus fitting into neither of these broad families of coping. As a result, the current thesis is more concerned with the instances of coping and how they are associated with adaptation to IBD, rather than the relationship between coping families or styles and adaptive outcomes that is popular in the chronic illness literature seemingly out of convenience. 32 1.6.3. Sense making Research on sense making has drawn from two key models: the Self-Regulatory Model (SRM) and Cognitive Adaptation Theory. The Self-Regulatory Model (SRM) In addition to coping, research has also addressed the process of living with a chronic condition with a focus on sense making which was first established by Leventhal et al.'s (1980) Self-Regulatory Model (SRM). The origins of this theory were borne by the likes of Leventhal et al.'s (1965), Leventhal and Singer (1966), Leventhal et al. (1967) and Leventhal and Trembly's (1968) efforts to understand the importance of fear and danger perceptions in affecting attitude towards acute threats and protective health actions against these threats. Ultimately, their focus on fear proved fruitless as fear appeared to only promote protective actions in the shorter term. Leventhal and colleagues therefore built on this model of fear communication and distress control by arguing that behavioural regulatory mechanisms were not governed merely by a valid and believable health danger along with an effective coping strategy to counteract the threat, but that the representations of danger that patients formulate themselves and the subsequent coping strategies that they develop based on what they believe to be the most appropriate means by which to deal with their own perception of the threat, also impact health behaviours. For example, early work by Leventhal and Cleary (1979) on smoking behaviour found that smokers switched to filter and low tar and low nicotine cigarettes because they seemed to be effective ways of coping with the health danger of smoking based on their own idea of the threat and what they perceived the best way to deal with this threat. Consequently, Leventhal et al. (1984) proposed their self-regulating processing system to explain how patients create their own common sense models about their illness which play an important role in influencing one's adjustment to illness. The SRM is 33 comprised of three hierarchical, interrelated and recursive stages, namely 'representation' of the illness experience that might guide 'coping', involving the planning and execution of health-related responses, culminating in 'appraisal' which involves the monitoring and evaluation of one's coping efforts. This sense making process may in turn lead to a number of results including the revision of one's evaluation criteria, the selection of new coping strategies and a change in representation of the health threat as well as a change in the health problem itself (Scharloo & Kaptein, 1997). Core to the SRM is the idea that representations of a health threat are a combination of lay beliefs about the threat together with existing schemata. It is primarily within the lay beliefs of the patient though that Leventhal and colleagues argue illness representations are constructed. In particular, these representations are composed of five key components: 'identity' - the label given to the condition and, in an attempt to legitimise it, its seemingly associated symptoms; 'cause' - the patient's ideas regarding the supposed cause of the health threat as they perceive it, based on personal experience as well as information accrued from a possible host of other sources; 'time line' the patient's belief about the duration of the illness, i.e. whether it is an acute or chronic threat; 'consequences' - the patient's beliefs about the effects and outcome of the health threat and the impact it will have on them physically and socially; 'curability/controllability' - the patient's beliefs about whether the condition can be cured or kept under control and the degree to which the patient can influence this (Hale et al., 2007). The SRM has been applied to a vast number of chronic illnesses and thus the evidence base from which to draw conclusions as to the efficacy of the relationship between illness perceptions and illness outcomes is abundant. Early support for the SRM was reported by the likes of Meyer et al. (1985) who assessed the SRM in the context of patients with hypertension. In this study, illness representations were assessed in relation to their impact on adherence to blood pressure medication and the likelihood of patients dropping out of 34 treatment. With regards to adherence to medication, 46 out of 50 patients in a subgroup of 'continuing treatment' identified symptoms of high blood pressure, with 17 of the 46 believing that treatment would influence their blood pressure and modify their symptoms and 12 of these 17 taking their medication as prescribed, with a further nine of these 17 displaying good control of their blood pressure. Five of the 17 who did not adhere to their medication as prescribed treated their blood pressure as though it were an acute illness and thus only took medication when they believed their symptoms were present. In contrast, medication was adhered to by only nine of the remaining 29 patients who believed treatment did not affect their symptoms, and of these only seven of 29 had good control of their blood pressure. In this instance, those demonstrating stronger illness representations of identity and controllability were more adaptive in their illness behaviour in the form of adhering to their medication and this resulted in generally better health outcomes for these patients. Meanwhile, for the 'newly treated' subgroup of patients, both symptoms and timeline were related to decisions to enter and remain in treatment. When the initial interview was used to divide the 'newly treated' patients into those who did and did not identify symptoms with their high blood pressure, a greater percentage of those who reported symptoms had dropped out of treatment by six month follow-up. Upon further examination, unexpectedly, 23 of the 46 who identified a symptom with blood pressure indicated that they had specifically mentioned that symptom on their first visit to their doctor. When this group of 23 patients was compared to the 42 who did not say they reported symptoms to their doctors, it was found that 14 of the 23 from the former had dropped out of treatment, compared with 10 of the 42 from the latter. Therefore, newly treated patients who said they told their doctor about their high blood pressure symptoms were more likely to drop out, due apparently to their interpretation that the disappearance of symptoms represented a cure of the disease and the continued presence of symptoms as a failure in the treatment. With regards to the effects of the perceived 35 timeline of the illness, 'newly treated' patients who saw their problem as chronic (18 of 65) had a low proportion of drop outs over the next six months (3 of 18). Meanwhile, those who saw their problem as cyclic or episodic (21 of 65) had a higher dropout rate (6 of 21), while those who saw the problem as acute (26 of 65) had the highest dropout rate (15 of 26). As such, this provided evidence for the timeline aspect of illness representations as being a factor in determining whether patients are compliant with treatment and thus whether they adapt appropriately to their illness. More recent applications of the SRM have included the work of Moss-Morris et al. (1996), among others. In their study on 233 CFS sufferers, it was found that the illness perception components of illness identity, emotional causes, controllability and consequences had the strongest overall association with adjustment such that participants with a strong illness identity and thus attributed certain symptoms to their illness, who believed their illness was out of their control, caused by stress and had very strong consequences were most disabled by their illness and psychologically impacted. Furthermore, a series of regression analyses indicated that these illness perceptions contributed to a greater percentage of the variance in levels of disability and psychological wellbeing than did the coping strategies used by the participants to manage their illness, such as venting emotions and methods of disengagement from the illness. Knibb & Horton (2008) meanwhile assessed the importance of illness perceptions on 156 allergy sufferers, finding that illness perceptions explained between six and 26% of the variance on measures of psychological distress in multiple regression analyses. Specifically, a strong illness identity and emotional representations were associated with a higher level of psychological distress, while strong personal control beliefs were associated with lower levels of distress. Coping in this instance explained between 12 and 25% of the variance on measures of psychological distress, and while adaptive coping strategies such as positive reinterpretation and growth did partially mediate the link between 36 illness perceptions and distress, illness identity, emotional representations and personal control retained significant independent associations with distress. Others such as Arat et al. (2011) have applied Leventhal's SRM to systemic sclerosis, finding that in a study of 217 patients, poor physical health was significantly associated with the perception of serious consequences and a strong illness identity, while good mental health was significantly associated with low illness identity scores and low emotional response to the illness. Once again, coping variables were less significantly correlated with physical and mental health compared with illness perception items. In addition, another finding of interest was that illness representations contributed more than the characteristics of the patients' disease such as disease activity and other severity parameters. These findings thus supported the importance of illness representations in the SRM in predicting positive health outcomes over and above not only other measures of patient cognitions and coping but also biomedical markers of disease severity. Meanwhile, Norton et al.'s (2014) study of 277 rheumatoid arthritis patients found that those patients characterised by a negative representation of their illness attributed more symptoms to their condition and reported stronger perceptions of consequences, chronicity and cyclicality of their condition as well as lower perceived control compared to the positive representation group. Furthermore, the members of the negative representation group were more likely to have higher levels of pain and functional disability as well as, longitudinally, increased levels of pain, functional disability and distress. Cognitive Adaptation Theory (CAT) Another perspective on sense making in chronic illness was put forward by Shelley Taylor in her Cognitive Adaptation Theory (CAT; Taylor, 1983). Taylor's 'CAT' describes the ability for individuals to overcome and readjust in the face of threatening personal events and was coined on the back of the discovery of this phenomenon in work with cancer patients, cardiac patients, rape victims and other individuals facing life-threatening events. 37 CAT dictates that this readjustment process is predicated on three core themes. The first is searching for 'meaning' in the midst of the experience in the form of a search for an answer as to the significance of the event in question. Meaning can also be determined by associating attribution and thus identifying the cause of the event. It is these attributions in particular that are crucial in the search for meaning according to CAT. Heider (1958) previously described how people are all essentially naive psychologists and we all attempt to make sense of our environment and the actions that occur within it, even finding supposed causal attributions when there is no factual basis for these attributions. Heider argued that we make these decisions on the cause of events in the search for meaning based on either internal attributions such as personality or belief characteristics of the person at the heart of the event, or external attributions, i.e. characteristics of our environment or situation in which the threatening event occurred. In the case of CAT, Taylor et al.'s (1984) seminary work on the causal attributions of 78 women with breast cancer found that these patients' beliefs about the cause of their cancer ranged from general stress (41%), carcinogens (32%) and diet (17%), to those who believed their cancer was borne from a blow to the breast such as a car accident or fall (10%). Crucially, what was found was that no specific attribution type was associated with adjustment to a greater degree than any other, thus causal attributions in general are important rather than the specific type of attribution, with adjustment in this instance being operationalised by an amalgamation of scores from the Global Adjustment to Illness Scale (GAIS) (Derogatis, 1975) as measured by the physician; the interviewer's independent rating on the GAIS; the patient's self-rated adjustment on a 5 point scale; patient self-reports on a number of psychological symptoms such as anxiety and depression; the patient's score on the Profile of Mood States (McNair & Lorr, 1964); and the Index of Well-Being (Campbell et al., 1976). As well as searching for meaning in one's life in the face of a threatening event through these causal attributions though, according to CAT, people also search for meaning 38 by endeavouring to understand what their life means in light of the threatening event. In this instance, psychological adjustment was found to be associated with patients who were able to frame their cancer diagnosis in a positive manner and use the experience as a reason to garner more satisfaction and meaningfulness from life. The second core theme is that of 'mastery' which is concerned with regaining control over life in the face of a threatening event. Mastery can be ascertained by the person reflecting on what they can do to deal with the event in the present moment and what they feel they can do to prevent the event from reoccurring in the future. In the sample of cancer patients referred to thus far, two thirds of those interviewed believed they had at least a certain degree of control over the course of or recurrence of their diagnosis, while just over a third of patients believed they had a lot of control over their cancer and the remaining patients believing that while they perceived themselves as having no personal control over the cancer, it could be controlled by their doctor and the treatments they prescribed. In terms of the efficacy of this belief in personal control or the belief in the ability of physicians and treatments to establish control over the disease, these cognitions were strongly associated with overall positive adjustment, with the combination of the two beliefs proving to be even more suggestive of positive adjustment. Alternatively, some patients adopted behavioural techniques in an effort to both reduce the likelihood of their cancer recurring as well as establishing control over their cancer at the current time such as changing diet or eliminating certain medications like birth control pills. Others meanwhile actively sought to be more knowledgeable about their cancer by acquiring information so as to then also attempt to establish some control over it, while others still attempted to control the side effects of their medication using both behavioural techniques, such as sleep or other medications, and cognitive techniques such as distraction or meditation. These findings were supported by literature such as that of Thompson (1983) who postulated that the repercussions of averse 39 stimuli, such as the stress that is likely to occur in the face of a threatening event like the cancer diagnoses being investigated by Taylor et al., can be lessened by establishing control, be it behaviourally, cognitively, retrospectively, or by amassing information on the event, largely corroborating Taylor's conclusions on the importance of mastery. The final aspect of cognitive adaptation according to Taylor's CAT is that of 'selfenhancement' which Taylor argues is rather focused on social comparisons than specific cognitions as the former two themes were mostly characterised by. In carrying out these social comparisons, a person facing a threatening event creates what has been termed 'positive illusions' so that they may begin to feel good about themselves and recover the selfesteem that is often lost when one is a victim of a threatening event, even when the event can be attributed to external circumstances. This notion of socially comparing oneself to boost self-regard was first posited by Festinger (1954) in his theory of social comparison processes whereby he built upon his seminal paper on how social communications can promote opinion conformity and subsequent uniformity within groups (Festinger, 1950) to go on to explain how people are driven to form opinions of themselves by comparing their abilities and opinions with others around them. Festinger proposed that these social comparisons can be 'upward' or 'downward' depending on if one compares themselves to others perceived as doing better or by comparing to someone they perceive as worse off, respectively. In the context of Taylor et al.'s cancer sample, what was found with regards to social comparisons was that almost all the patients felt that they were doing as well as if not somewhat better than other women facing the same predicament, in other words almost unanimously the patients in the sample made downwards comparisons in an attempt to bolster their selfesteem. Specifically, however, these social comparisons can differ slightly in that while some are efforts at validating one's favourable image of oneself, sometimes people may demonstrate cognitive adaptation and self-enhance by socially comparing in order to 40 construct self-impressions also. In these instances, people will compare against those who are indeed worse off but perhaps also coping well in order to draw inspiration and to act as a guide for future behaviour, with patients in the cancer group in question for example comparing themselves with women who had metastatic cancer or who had undergone double mastectomies. CAT has been utilised extensively in the adjustment literature to date, being applied in particular by Helgeson, the first such application taking place in a study which explored the applicability of CAT in predicting adjustment in a sample of heart disease patients following coronary angioplasty (Helgeson, 1999). A total of 278 male and female participants were recruited who had been treated for a coronary event with percutaneous transluminal coronary angioplasty and partook in an interview while they were in hospital and then six months later. The primary finding was that indicators of the three CAT core themes generally predicted positive adjustment among the patients. In conjunction with this, there was found to be strong robustness of the CAT-related cognitions among patients, suggesting they were consistent in the face of recurrent coronary events. Helgeson and Fritz (1999) then followed this up by assessing which of the components of CAT would predict new coronary events after a first coronary angioplasty, with their results indicating that patients who respond to their illness by perceiving control over their future, by having positive expectations about their future, and by holding a positive view of themselves seemed to be less at risk of developing a new cardiac event after a first coronary angioplasty. Helgeson (2003) then built on this study with an investigation on 298 male and female participants who were four years removed from coronary angioplasty surgery using the Cognitive Adaptation Theory Index (CATI), developed during hospitalisation for the patients' initial angioplasty procedure and which assesses the degree to which one positively views oneself, has a positive view of one's future, and the degree of personal control one has over their daily life. Once again CAT, as 41 operationalised by the CATI, predicted positive adjustment to disease, as well as a reduced likelihood of sustaining a subsequent cardiac event over the 4 year period. More recently, Helgeson et al. (2014) found similarly encouraging results when assessing whether CATI scores for youths with type 1 diabetes were predictive of emerging adulthood outcomes. In this study of 118 youths with (n=118) and without type 1 diabetes (n=122), CATI at baseline during the participants' senior year of high school predicted reduced psychological distress, enhanced psychological wellbeing, increased friend support, reduced friend conflict, the presence of romantic relationships, reduced likelihood of romantic breakups, higher GPA, higher work satisfaction, and lower work stress during the transition to emerging adulthood, with stronger findings in these domains for those with rather than those without diabetes, indicating the importance of cognitive adaptation for those with chronic illness in particular. Also of significance is the finding that the implications of CAT appeared to extend beyond merely psychological adjustment to relationship, vocational and diabetes-related outcomes in this instance. In addition, CAT has been further validated by its application to late-stage cancer patients by Christianson et al. (2013). In this study of 80 patients with varying cancer diagnoses, findings suggested that higher scores on questionnaires measuring the three core CAT constructs, namely greater self-esteem, control and meaning, predicted physical and psychological quality of life, with physical quality of life particularly influenced by control beliefs and psychological quality of life particularly influenced by self-esteem. Meanwhile, Czajkowska et al.'s (2013) study on patients with nonmelanoma skin cancer also corroborates the validity of CAT, finding that in a sample of 57 patients, the CATI successfully predicted participants' distress, accounting for 60% of the variance and lending support for the efficacy of Taylor's CAT constructs in being attributable with adjustment to chronic illness. However, not all studies have produced results that are entirely unanimous in their support for CAT and its ability to predict positive adjustment. For example, Moore et al. (2006) found in a study of 42 123 patients with venous thromboembolic disease that, in line with much of the aforementioned literature, the CAT constructs of mastery and self-esteem were associated with positive adjustment to disease. In contrast to much of the previous studies however, the construct of meaning was associated with elevated levels of distress for these patients. Similar limitations of the theory were demonstrated by Tomich and Helgeson (2006) who looked into the effect of the components of CAT on QoL and benefit finding in breast cancer patients. They found in this instance that baseline perceptions of personal control over illness, but not general self-esteem or optimism, were associated with patients' reports of worse physical functioning, worse mental functioning, and less benefit finding 5 years later for recurrent women but not disease-free women, raising questions over the importance of perceived control on adaptation. 1.6.4 Criticisms of the SRM and CAT Both the SRM and CAT share a common theme in the form of the prism through which they conceptualise adjustment to illness. They are predicated on the way in which cognitions have an impact on illness outcome and it is this aspect of the theories that lends itself to their main criticism: it is highly likely that other factors influence the proposed pathways of these models from cognitions to outcome in the case of CAT, and from cognitions to outcome via coping in the case of the SRM. For example, neither model accounts for the importance of social interactions and support in the management and outcome of illnesses. Such social factors have time and time again been reported in the literature as having an impact on aspects such as medication adherence in diabetes (Miller & DiMatteo, 2013), influencing adaptive or maladaptive coping which in turn influenced pain levels in patients with rheumatoid arthritis (Holtzman et al., 2004), as well as being associated with impairment in multiple sclerosis (Rommer et al. 2016). Further to this, behavioural factors in CAT are indirectly alluded to in terms of the core theme of mastery 43 and its focus on a patient's belief in their ability to carry out actions to overcome and prevent further threat, but the theory is centred on the importance of cognitions as reflected by the theory's name. As for behavioural factors in the SRM and how they influence adjustment, they are in this instance seen as a mediator of the main focus of the theory, the illness representations, and subsequent illness outcomes. However, as Hale et al. (2007) point out, illness representations may be associated with outcomes relatively independent of the coping strategies used. For example, research into the buffering effects of coping styles against stress in rheumatoid arthritis found that an avoidant coping style did not appear to be influenced by perceiving that the diagnosis had serious consequences (Treharne et al., 2007). In conjunction with this, as outlined in this introduction, while the relationship between the processes of coping and sense making and the outcome of QOL is likely iterative, the same can be said of the different components of the SRM. For example, the belief that an illness has no serious consequences could be deemed a coping strategy such as denial just as much as it is deemed an illness cognition by Leventhal et al (Ogden, 2004). 1.7 Outcomes of a chronic condition The key outcome assessed in the context of chronic conditions is Quality of Life (QoL). As seen with the psychological processes thus far, particularly coping, QoL is similarly complex in its definition and conceptualisation due to its subjective nature. Liu (1976) for example argued that it was possible to define QoL in as many ways as there are people, due to the differing ways in which individuals perceive the notion of quality in their life. Indeed, the context by which the concept of QoL is applied can again change its meaning, being used to assess aspects of societal or community wellbeing as well as individuals or groups (Felce & Perry, 1995). The breadth of the term is typified by the World Health Organisation's (1997) who define QoL as 'an individual's perception of their position 44 in life in the context of the culture and value systems in which they live and in relation to their goals, expectations, standards and concerns'. It is a broad ranging concept affected in a complex way by the person's physical health, psychological state, personal beliefs, social relationships and their relationship to salient features of their environment'. Flanagan (1982) became one of the first researchers to endeavour to take this conceptualisation of QoL and operationalise it, developing the Quality of Life Scale (QOLS). This was a 15 item instrument that measured five conceptual domains of quality of life: material and physical wellbeing, relationships with other people, social, community and civic activities, personal development and fulfilment, and recreation. The QOLS was then adapted slightly for use with chronically ill patients by adding one item on the degree to which one felt they were able to maintain their independence. The measure has since then been applied to patients from a variety of chronic illness populations to assess their perceived quality of life including diabetes (Burckhardt et al., 1989), osteoarthritis (Blixen & Kippes, 1999), irritable bowel syndrome (Sperber et al., 1999), systemic lupus erythematosus (Abu-Shakra et al., 1999) and chronic obstructive pulmonary disease (Anderson, 1995). However, despite the purported efficacy of the QOLS as a reliable and valid instrument for measuring quality of life from the perspective of the patient (Burckhardt & Anderson, 2003), measuring QoL in patients is increasingly becoming specific to illness groups with more focus being placed on health-related quality of life (HRQoL). HRQoL is a multidimensional construct consisting of at least three broad domains physical, psychological and social functioning, that are affected by one's disease and treatment (Megari, 2013), rather than simply the QoL of a patient or patient population. Furthermore, while generic instruments for measuring QoL in chronic illnesses such as the aforementioned QOLS or the more recent SF-36 (Ware et al., 1993) or EuroQol (The EuroQol Group, 1990) do allow for broad comparisons across conditions due to only one 45 metric being used and then generalised amongst other disease populations, disease-specific instruments tend to provide greater precision and sensitivity to clinically important changes that are unique to the disease in question and thus possibly missed by broader assessment methods (Staquet et al., 1998). Examples of such disease-specific measures of QoL include the likes of the Diabetes-39 (Watkins & Connell, 2004), the MG-QOL (Mullins et al., 2008) which was developed to be used with patients suffering from a rare, autoimmune disorder related to muscle weakening known as Myasthenia Gravis, as well as the IBDQ (Guyatt et al, 1989) for those with IBD. As such, it is indeed the IBDQ tool that will be utilised later in the thesis when measuring QoL for the current sample of patients with IBD. This particular measure assesses QoL in IBD by accounting for four domains of the patient experience: systemic symptoms, bowel symptoms, social factors and emotional functioning and has demonstrated excellent reliability and validity (Pallis et al., 2004). While these types of multidimensional measures of health status have been criticised for being too broad and therefore defining QoL in vague terms, it was felt that for the purpose of this thesis a multidimensional outcome measure was not only more pragmatic than a unidimensional measure of health, but also is representative of the ideally generalised benefit of being adapted to IBD. Examples of alternative unidimensional measures include the 'hospital anxiety and depression scale' (HAD) (Zigmond & Snaith, 1983) which focuses on mood or the 'McGill pain questionnaire' (Melzack, 1975) which assesses pain levels which, while being effective at measuring one particular aspect of health, would likely need to be used in conjunction with a host of other measures to get a global view of the outcomes associated with adaptation. In conjunction with this, while measures of health status such as the IBDQ are subjective in their nature due to being self-report, the decision to employ this type of measure of outcome is guided by the perspective one takes when assessing health status. Those concerned with biomedicine may for example view health status as the presence 46 or absence of death in which case mortality rates, an objective measure of health status, are a more appropriate assessment tool, whereas the current thesis is concerned with health psychology and thus health status in this context is more complex and requires a greater variety of outcomes to be accounted for. Not only this, but so as to be in line with a health psychology model of what health means, individuals themselves should rate their perceived health status (Ogden, 2004) and as such the IBDQ is again an appropriate outcome measure of adaptation for the current thesis. Research into chronic illness is therefore commonly associated with either objective or subjective measures of health status. Furthermore, while clinicians and other biomedical health professionals are traditionally focused on the evaluation of symptoms of disease and functional status of the patients, patients are more often focused on the complete range of health status (Rumsfeld, 2002) which can be reflected in measures of QoL, and HRQoL in particular. 1.8 The working model of adjustment to chronic illness – integrating both processes and outcomes Research exploring the experiences of those with chronic illnesses have therefore focused on both the process of having an illness, highlighting the role of coping and sense making and the outcomes with a focus on quality of life. Some research has also drawn upon comprehensive models of chronic illness which address both of these issues. This thesis will focus on one key model by Moss-Morris (2013). In an attempt to overcome some of the issues thus far mentioned in terms of building on the conceptualisation of adjustment, Moss-Morris (2013) developed a working model of adjustment to chronic illness. This model posits, firstly, that multiple background factors should be taken into account when we assess how people respond and ultimately adapt to 47 various illness stressors. These background factors can come in the form of personal factors such as early life experiences, values and life goals, as well as demographics, or in terms of social and environmental factors such as one's socioeconomic background, the availability of health and social care, as well as the degree of social support one receives. Meanwhile, MossMorris argues that illness-specific factors such as the nature of one's symptoms, the degree to which these symptoms are disabling or disfiguring, the prognosis associated with the illness and the treatment regimen required to attempt to control the illness and its associated side effects then in turn determine, firstly, the nature of possible key critical events, as well as possible ongoing illness stressors. Examples of the former include ascertaining the potential threat to one's mortality, the possibility of illness relapse and progression, as well as its ability to manipulate one's identity and life roles, while examples of the latter include one's ability to manage social relationships and relations with health professionals, uncertainty regarding one's future, determining one's limits in light of the illness, the struggle to maintain autonomy, as well as managing ongoing treatment and potential lifestyle changes. These illness events and ongoing stressors culminate in the emotional equilibrium of the patient being upended. Adjustment to chronic illness, therefore, is proposed in this model as the process of returning and maintaining equilibrium in the face of these chronic stressors, not dissimilar to the alluded to notion of returning to equilibrium following a health threat in the SRM (Leventhal et al., 1997). Moss-Morris goes on to claim that this target of equilibrium can be broken down into psychological, social and physical adjustment, incorporating aspects such as, among other things, managing levels of distress experienced, limiting the amount of interference the illness causes on one's life roles and relationships, one's ability to manage the illness, as well as retaining positive affect. Based on this conceptualisation, disequilibrium, or maladjustment, persists when there is ongoing disproportionate distress, interference of life roles and relationships, poor illness management and low positive affect. Conversely, 48 successful adjustment is accordingly determined by low perceived levels of distress, low interference on life roles and relationships, good illness management and high positive affect. In order to achieve this successful adjustment and return to equilibrium, or fail to achieve adjust and experience ongoing disequilibrium as the case may be, Moss-Morris argues that in conjunction with the previously described background factors of the patient in question, there are certain cognitive and behavioural strategies that individuals commonly utilise to manage illness stressors. For example, cognitive factors often cited in the context of successful adjustment include possessing a sense of control with regards to management of the disease, self-efficacy regarding generic life situations, an ability to benefit find and positively reinterpret situations, an acceptance of one's illness, as well as high perceived social support; while poor adjustment in this domain is often associated with high perceived stress, negative symptom interpretation, dysfunctional cognitions such as catastrophising, helplessness, as well as suppressing negative affect. Behavioural factors associated with successful adjustment meanwhile include coping by using problem-focused strategies and seeking social support, engagement in positive health behaviours, adherence to medical and selfmanagement regimes, maintaining activity levels in the face of illness and appropriately expressing emotions. Alternatively, adjustment difficulties are often associated with behavioural factors such as coping through avoidance, unhelpful responses to symptoms such as consistently reducing activity, resting and hyper vigilance of symptoms, as well as venting or repressing emotions. Crucially though, Moss-Morris asserts that these cognitive and behavioural factors need to be determined empirically for each illness group, further distancing the current model from previously discussed models of understanding adjustment to health threats in that the previous paradigms provide a basic framework for our understanding of the concept but they are not specific to any particular illness and do not approach the construct in a multifaceted, patient-centric manner. As such, CAT and 49 Leventhal’s SRM undoubtedly provide frameworks from which we can understand adaptation to chronic illness, but to fully comprehend adaptation we need to also account for the nuances of the lived experience of the illnesses so as to reflect the biopsychosocial nature of chronic illness. 1.9 The central role of adaptation While thus far we have seen that the literature often refers to the process of adjustment rather than adaptation, it is the term adaptation that the current thesis will be focused on going forward. Primarily, the reasoning for this is that the concept of adaptation cuts across the key psychological processes that we have discussed up to this point. Throughout the individual notions of Lazarus' coping paradigm, Taylor's CAT, Leventhal et al.'s SRM and Moss-Morris' working model of adjustment to chronic illness is a seemingly transcendent theme of adaptation that binds these aforementioned concepts under one overarching term. Secondly, while the literature seemingly deems adjustment and adaptation as interchangeable terms, the term adaptation more accurately reflects the life-changing nature of chronic illness, and IBD in particular, and the subsequent need for patients to constantly react and evolve in the face of dynamic disease circumstances and evaluate whether they are adapted in the present moment and, if not, how one can achieve this goal of adaptation. This will be discussed in more detail in Chapter 3. Within the chronic illness literature there have indeed been numerous attempts at bringing together the notions of illness representations, sense making and coping alongside social and behavioural factors in an attempt to understand adaptation in a multifaceted manner. These studies have also endeavoured to demonstrate the impact of these biopsychosocial factors that affect adaptation on a range of outcomes. For example, Dennison et al. (2009) and Dennison et al. (2010) conducted extensive work on the psychological, 50 social and behavioural correlates of adjustment in patients with MS, finding that the outcome of distress was correlated with unhelpful beliefs about the self, unhelpful beliefs about emotions, acceptance, and unhelpful cognitive responses to symptoms and illness perceptions, while social support and interactions with others and certain health behaviours such as behavioural responses to symptoms were also associated with adjustment outcomes. Similarly, McCathie et al. (2002) found that higher levels of catastrophic withdrawal coping strategies and lower levels of self-efficacy of symptom management were associated with higher levels of depression, anxiety and reduced QoL, while higher levels of positive social support were linked to lower levels of depression, and anxiety and higher levels of negative social support were linked to higher levels of depression and anxiety in patients with chronic obstructive pulmonary disease. However, these studies do not fully conceptualise adaptation to their respective illnesses as such, rather they are conceptualised as a product of the measures of adaptation that the researchers believed were most appropriate or were particularly interested in associating with outcomes. For example, Dennison et al. (2010) measured participants on The Psychological Vulnerability Scale, The Beliefs and Emotions Scale, The Acceptance of Chronic Health Conditions scale, The Brief Illness Perception Questionnaire and The Cognitive and Behavioural Responses to Symptoms Questionnaire as 'potential predictors of adjustment', while McCathie et al. (2002) selected The Coping with Illness Questionnaire, The COPD Self-Efficacy Scale and The Illness-Specific Social Support Scale as their proxy measures of adjustment or adaptation. 1.10 Process, outcomes and adaptation in IBD While the CAT and SRM have not been as extensively applied to IBD as they have some other chronic illnesses, Rochelle and Fidler's (2013) application of the SRM is one of the few opportunities to observe the impact of illness representations on outcomes in this patient population. In this instance, illness representations were investigated in relation to the 51 outcomes of quality of life (QoL), anxiety and depression. With regards to QoL, those who believed their illness would last a short time were more likely to have higher QoL scores, while patients who believed their IBD would have serious consequences displayed lower QoL scores, as were those patients who did not perceive themselves to have control over their illness. In terms of anxiety, analyses suggested that patients who perceived their illness to have serious consequences were more likely to have higher levels of anxiety, as well as those who perceived themselves to have less control over their illness and, somewhat paradoxically, those who demonstrated better understanding of their illness. Finally, in terms of predictors of depression, this was found to be particularly associated with those patients who believed that their disease would last a long time, as well as those who did not perceive that medication would control their illness and, again perhaps surprisingly, those who had a better understanding of their illness. The subsequent conclusion we can draw from these results by Rochelle and Fidler (2013) therefore is that sense making in the form of illness representations are indeed also important in IBD as was echoed in all of the aforementioned chronic illness literature, having an impact on the psychological status of patients with IBD, which in turn has ramifications for how these patients cope, with those who perceived their illness to have serious consequences, for example, being more likely to demonstrate maladaptive coping in terms of expressing their emotions. These behaviours in turn then have ramifications on QoL and the psychological adjustment of patients, which may then indeed augment disease-related impacts which in turn can further perpetuate the cycle of negative outcomes. However, Leventhal et al.'s proposed pathway from illness representations to outcomes via coping was disputed by Dorrian et al. (2009) who found that although illness perceptions, and particularly the perception of consequences of IBD, was consistently associated with the adjustment outcomes of psychological distress, QoL and functional 52 independence, coping did not significantly add to predicting adjustment once illness perceptions were controlled for. Indeed, of all the psychological processes discussed, the coping literature and has garnered the most attention in the IBD literature. As stated previously, much like other research has concluded, emotion-focused, avoidance-based strategies have been found to be most predictive of poor outcomes, being linked, for example, to increased psychiatric distress and poorer QoL (van der Zaag-Loonen et al., 2004; Jones et al., 2006). These results have been echoed by Voth and Sirois (2009) who found that avoidant coping was associated with poor adjustment, with adjustment in this case being operationalised as a product of coping efficacy, acceptance and helplessness indices. However, as outlined earlier, when one assesses emotion-focused coping in the context of coping instances such as emotional modulation and acceptance, then emotion-focused coping has been found to be associated with the outcomes of fewer somatic complaints and symptoms of anxiety and depression (Thomsen et al., 2002). The relationship between problem-focused coping and outcomes are similarly unclear. Pellissier et al. (2010) found, for example, that problem-focused coping was associated with better psychological outcomes such as anxiety, depressive symptomatolgy and negative mood; whereas the likes of Petrak et al. (2001) found that problem-focused, or 'active' coping as it was termed in this study, was associated with poor HRQoL. Others still failed to find any link at all between relationship between QoL and coping (Larsson et al., 2008; Iglesias-Rey et al., 2014), suggesting there are some inconsistencies in the impact of particular coping strategies on outcomes in IBD. Attempts at understanding adaptation in the IBD literature meanwhile are lacking in terms of both its conceptualisation and operationalisation. While Devlen et al.'s (2014) conceptual model of the impacts of inflammatory bowel disease has contributed to painting a somewhat complete picture of the biopsychosocial nature of IBD, which was previously 53 argued as essential if we are to develop an understanding of adaptation to IBD according to Moss-Morris' (2013) working model, they fail to explain what the process of adapting to these challenges entails. Devlen et al.'s model depicts the interconnectedness of patients’ bowel and systemic symptoms with not only the psychological impacts of the disease, as well as the impacts that come about as a result of treatment, relationship difficulties, social and leisure activity and work, school and parenting limitations. Over time, multiple researchers have endeavoured to fill the gaps on what aspects help patients overcome and adapt to these biopsychosocial impacts. Opinions on what exactly constitutes this adaptation have ranged from Olbrisch and Ziegler's (1982) finding that a patient's general level of self-esteem and social competence was most closely associated with disease-related adjustment, while Maunder and Esplen (1999) developed a model of normal adjustment to IBD through a synthesis of existing literature and clinical experience in a tertiary care medical and surgical IBD centre, coming to the conclusion that normal adjustment could be understood as a process involving the interaction of three adaptive challenges: illness uncertainty, loss and change, and suffering. Voth and Sirois (2009) meanwhile have argued that poor adjustment to IBD was a product of attributions of self-blame which were directly related to increased avoidant coping. However, these studies were again based on arbitrary hypotheses of adaptation rather than developed from the bottom up. A relatively recent qualitative study by McCormick et al. (2012) meanwhile alluded to the fact that patients with IBD themselves believed in the importance of adaptations in the form of 'learning survival skills' such as maintaining balance of one's life in the face of numerous IBD stressors, being careful with what you eat, bathroom mapping and avoiding social stigma, as well as the importance of a resilient attitude in order to prevent their condition from interfering with opportunities in their life. While this study does breach the topic of adaptation to IBD, it requires further investigation to fully conceptualise. 54 1.11 Overview of aims for thesis The motivation for the current thesis is to attempt to consolidate the key psychological principles of the likes of CAT and the SRM with the more holistic approach of Moss-Morris' working model in the hope of understanding, firstly, what adaptation means specifically for patients with IBD, taking a bottom up approach to understanding the concept. The thesis then aims to build on this by investigating how this conceptualisation of adaptation can be operationalised and measured. This will be followed by an assessment of whether this framework of adaptation to IBD is potentially beneficial with regards to healthcare protocols in terms of whether or not it predicts the outcome of patients' subsequent QoL as measured by the IBDQ. Finally, upon understanding adaptation to IBD and what those who score high in adaptation do differently to those who score low in adaptation, the current thesis seeks to better understand how these high scorers were able to achieve becoming adapted to their illness, and likewise what factors are seemingly preventing the low scorers from becoming better adapted. In striving to reach these goals it is thus hoped that we may come some way towards resolving the long-standing issue of reconciling the interests of those primarily concerned with the psychological underpinning of disease and adaptation to it, with the interests of the patients who are at the epicentre of the issue and for whom adaptation presents a challenge on a plethora of fronts, not merely psychologically. Study 1: Patients' experiences of living with IBD: a qualitative study Aims: To explore and conceptualise the notion of adaptation in patients with IBD with regards to emotional, cognitive, social and behavioural aspects. This will provide a framework for subsequent operationalisation of the concept. Method: Qualitative study utilising in-depth, semi-structured telephone interviews with patients with IBD recruited from an opportunity sample of patients using social networking 55 sites and online IBD support groups. An interview schedule will be used to determine, firstly, the participants' history with IBD, then investigate how the participant felt, thought and behaved in reaction to their diagnosis and how, if at all, this differs to how they feel, think and behave with regards to their illness at the time of the interview. By looking at the contrast of the patients' cognitive, emotional, social and behavioural patterns between the two time points it is hoped that we may gain an insight as to how the patients adapted to the demands of their illness over time. Study 2: The development of a new measure to assess adaptation in patients with IBD and its utility in predicting QoL: a cross-sectional study Aims: This study seeks to, firstly, operationalise adaptation in patients with IBD through the formulation of a measure of adaptation, building on the previous study's conceptualisation of the construct. This study also aims to assess the degree to which the resulting measure of adaptation to IBD is associated with the concepts of sense making (as measured by the Brief Illness Perception Questionnaire; BIPQ) and QoL (as measured by the Inflammatory Bowel Disease Questionnaire; IBDQ). Finally, the study aims to utilise the adaptation measure to determine the degree to which patients' adaptation to IBD scores are predictive of their QoL scores as measured by the IBDQ. Method: Quantitative, cross-sectional design with recruitment again taking place from an opportunity sample of patients using social networking sites and online IBD support groups. The initial adaptation to IBD questionnaire will contain 40 items that will be derived from the results of Study 1 and subsequently refined through a factor analysis and tested for reliability using Cronbach's alphas. Participants will also complete the BIPQ and IBDQ, the scores of which will be correlated with those from the adaptation questionnaire to ascertain the uniqueness of the latter construct. Finally, participants’ adaptation to IBD scores will be 56 formulated and used to predict QoL scores in a multiple regression analysis. All questionnaires will be completed online through Qualtrics. Study 3: Patients' explanations for their degree of adaptation: a qualitative study with a quantitative component Aim: To explore the differences between those participants scoring in the top and bottom 25% of scorers on the adaptation to IBD questionnaire on the key aspects that these patients felt encouraged or inhibited, respectively, their adaptation to IBD. The aim of this is to then understand better what aspects can be manipulated to assist patients with IBD to score higher on the key factors of adaptation as put forward by the adaptation to IBD questionnaire. To also explore whether adaptation changes over time. Method: Qualitative study employing in depth, semi-structured telephone interviews with an opportunity sample of participants scoring in the top and bottom 25% of scorers on the adaptation to IBD questionnaire. An interview schedule will be used to explore what psychological, social and behavioural factors the patient felt contributed to their score for each of the adaptation to IBD questionnaire factors. Further, patients completed the A-IBD between 10 and 16 months after their first assessment to explore changes over time. 57 Chapter Two Study 1: Patients' experiences of living with IBD: a qualitative study 2.1 Overview The previous chapter first reviewed the literature regarding the definition, prevalence, causes and consequences of IBD. It then explored the research regarding the key psychological constructs of processes and outcomes and how they have been applied to chronic illness in general as well as IBD, focusing on the notions of coping and sense making for the former and QoL for the latter. The aim of this chapter is to explore and conceptualise the notion of adaptation in patients with IBD using a qualitative study on the emotional, cognitive, social and behavioural changes that patients with IBD make, if any, over the course of their disease experience from diagnosis up until the time of interview. This study has been published: Matini, L., & Ogden, J. (2015). A qualitative study of patients’ experience of living with inflammatory bowel disease: A preliminary focus on the notion of adaptation. Journal of Health Psychology. doi: 10.1177/1359105315580463 2.2 Introduction Inflammatory Bowel Disease (IBD) is an umbrella term that encapsulates two similar yet ultimately distinct chronic diseases of the gastrointestinal tract: Ulcerative Colitis (UC affecting the innermost lining of the large intestine and the rectum) and Crohn’s Disease (CD - affecting any part of the digestive tract from mouth to anus and may permeate all layers of the bowel lining). The collective term is legitimised by the similarity in epidemiology and aetiology of the two conditions (Kirsner & Shorter, 1995). IBD usually develops during adolescence and is characterised by phases of acute abdominal pain and gut spasms, nausea, 58 fever, fatigue, diarrhoea and rectal bleeding (Gearry et al., 2006). Should patients manage to achieve periods of remission, fatigue still largely affects patients' daily functioning (CzuberDochan et al., 2013). The impact of IBD on the lives of patients is widespread. Besides having to deal with the aforementioned symptoms, patients often experience fear and humiliation as a result of stool incontinence for example, a profound effect on family life and work (Dudley-Brown, 1996), a feeling of losing control (Hall et al., 2005) as well as possible delayed growth and sexual development and social isolation (Sparacino, 1984). Compound these psychosocial effects of living with IBD with the common side effects of regularly prescribed medications, such as the steroidal treatment prednisolone which often includes weight gain and mood swings (Bayless, Drossman & Schrel, 1988), the high possibility of needing surgery or an ostomy bag (Canavan et al., 2006), as well as the increased risk of intestinal cancer in patients with IBD (Jess et al., 2005) and it becomes evident that it is almost impossible for quality of life to not be impacted in IBD (Larrson et al., 2008). Current theories on the cause of IBD are an amalgamation of infectious (Luther et al., 2010) environmental (Lakatos, 2009), genetic (Cho & Brant, 2011), and immunologic factors (Verhoef & Sutherland, 1990), which makes identifying a cure extremely difficult due to the numerous possible complex interactions that may trigger the illness. This issue is compounded by the growing incidence of IBD as demonstrated by Molodecky et al. (2012). Hanauer (2006) meanwhile has shown that the prevalence of IBD is also increasing in areas such as Asia, Africa and Latin America. It is thus imperative that health care practitioners better understand the lived experience of their patients and can be mindful of this when delivering treatment plans which may incorporate psychological interventions to maximise patients’ quality of life. This notion is further supported by the fact that although there have been major advancements in the treatment of IBD, in particular the rise of biologic agents 59 (Triantafillidis et al., 2011), the side effects of such medications can be harsh (Stallmach et al., 2010). The importance of patients’ health related quality of life has been highlighted by Drossman et al. (1989) who found that patients’ functional status and disease concerns were found to correlate better with scores of wellbeing and healthcare utilisation than a physician’s rating of disease activity. In conjunction with this, the presence of anxiety and depression amongst IBD patients regardless of disease severity (Vidal et al., 2008) further suggests the importance of developing an increased understanding of the lived experience in IBD. Psychological research to date has advanced the understanding of IBD and improved its prognosis. For example, Garrett et al. (1990) found that there was a relationship between daily stress and self-rated disease severity, while Sewitch et al.'s (2001) study on perceived stress revealed that, for those patients who experienced high levels of perceived stress, high satisfaction with social support decreased the level of psychological distress and facilitated adjustment to the disease. It is this notion of adjustment or adaptation however, that is often referred to in the IBD literature yet remains under studied. For example, Sajadinejad et al.’s (2012) review article on the psychological issues on IBD refers to the need for appropriate coping strategies and good adaptation in order to deal with the symptoms of IBD and its psychological impact, yet fails to define what good adaptation entails. This notion was also elaborated upon in McCormick et al.’s (2012) qualitative study on the experiences of patients with chronic gastrointestinal conditions where one of the main themes that emerged was that of ‘adaptations: learning survival skills’, but again no clear exploration of this notion was given. Therefore, the current study aims to address this gap in the literature by exploring the notion of adaptation in patients with IBD. In particular, the study focuses on their lived experience 60 from diagnosis to the present and observing whether there are adjustments in the ways in which these patients think, feel and behave with regards to their illness over time and, if so, what particular aspects of these domains change. 2.3 Method 2.3.1 Design A qualitative design with in-depth, semi-structured interviews. 2.3.2 Sample Participants were recruited via opportunity sampling from social networking sites, predominantly Twitter, as well as online IBD support forums by responding to a recruitment paragraph outlining the study. Participants were required to be at least 16 years of age, capable of providing informed consent, living in the UK and had been diagnosed with either CD or UC at least 18 months ago. Those UC participants who had undergone a colectomy and thus technically no longer suffered from the symptoms of UC were included as they still had to live with the effects and challenges of having surgery and it was felt this could still provide insight into the lived experience of IBD. 22 participants were recruited upon which point saturation was reached: CD (n=10 - five females, mean age=28.2 years, range=23-31 years; five males, mean age=30.2 years, range=18-39 years; mean time since diagnosis=8.8 years. All were recruited from Twitter), UC (n=12 - nine females, mean age=32.1 years, range=19-60 years; three males, mean age=42.7 years, range=36-48 years; mean time since diagnosis=5.0 years. Five recruited from Twitter, six from 'Colitis UK' forums, one from IBD forums on 'Daily Strength'). One prospective UC participant was not permitted to take part as they did not yet have a confirmed diagnosis. No participants who stated their interest or took part later 61 withdrew. All participants were given a pseudonym to ensure anonymity. Participant demographics are presented in Table 1 below. 62 Table 1: Participant demographics Pseudonym Sex Age (years) Diagnosis Time since diagnosis (months) Jessica F 23 CD 40 John M 30 CD 33 Elizabeth F 31 CD 102 Graham M 39 CD 276 Amy F 30 CD 46 Janet F 29 CD 81 Katie F 28 CD 228 Jason M 25 CD 91 Daniel M 39 CD 120 Brian M 18 CD 41 Jane F 26 UC 59 Jackie F 29 UC 74 Ian M 36 UC 46 Rebecca F 19 UC 34 Diane F 28 UC 34 David M 48 UC 81 Malcolm M 44 UC 44 Gemma F 41 UC 21 Megan F 21 UC 22 Chloe F 33 UC 46 Grace F 60 UC 178 Emily F 32 UC 86 63 2.3.3 Procedure Prospective participants were e-mailed an information sheet (See Appendix A) to describe the nature of the study along with an informed consent form (See Appendix B) that they were required to complete on their computer and return via e-mail to the principal investigator if they were willing to take part. All interviews were conducted in English, following a semi-structured interview schedule (See Appendix C) over the telephone at a mutually convenient time and were all carried out, audio taped, transcribed verbatim by the principal investigator and then stored securely on the University servers according to the Data Protection Act 1998. Telephone interviews enabled the recruitment of participants from multiple regions of the UK and also allowed the participants to divulge potentially embarrassing or intimate information from the safety of their homes and retain a degree of anonymity. Interviews lasted on average between 30 to 45 minutes and took place over the course of three weeks in March 2014. Once the interviews had been conducted, participants were granted the opportunity to ask any questions they had regarding their participation or to reflect on their experience. All participants were also e-mailed a debriefing form (See Appendix D) to explain the nature of the study. Ethical approval was obtained from the University Ethics Committee (See Appendix E). 2.3.4 Interview Schedule The same interview schedule was used for the CD and UC participants and was constructed by the authors. The first section of the interview covered the participants' history with IBD, focusing on the genesis of their illness (How did this diagnosis come about?), transitioning on to their early reactions to their diagnosis (What were your initial thoughts and feelings about your diagnosis?), and enquiring as to any initial coping techniques, ending with how participants are currently dealing with their illness (How do you currently feel about your 64 diagnosis of IBD? What factors have helped you manage your illness?) and a reflection on what the major changes have been over time, if any, with regards to how they manage their illness on an emotional, social, and behavioural level. The interview schedule was piloted with two patients with IBD prior to the study. 2.3.5 Data Analysis The interviews were analysed using thematic analysis as it provides a “flexible and useful research tool, which can potentially provide a rich and detailed, yet complex account of data” (p.78, Braun & Clarke, 2006). Within this analytical framework, an inductive, 'bottomup', approach was taken as our analysis was not based on any previous research into living with IBD, rather we sought to generate phenomenological data from which our themes could be developed. Furthermore, an essentialist theoretical perspective was chosen as our focus was on the experiences, meanings and reality of the participants. The interview transcriptions were each read through twice in order to become familiar with the data. Salient information that related to the lived experience of IBD was then coded and collated and sorted into a number of prospective themes that seemed to represent the data. Themes and subthemes were adjusted to include only those that could be supported by illustrative quotes from the interviews. 2.4 Results The participants described living with IBD in terms of three core themes, namely 'making sense of the illness', 'impact' and 'feelings'. Transcending these was an overarching theme of uncertainty which cut across all aspects of their experience. These themes will now be described with the use of illustrative quotes. They are then finally discussed within the context of the notion of 'the new normal'. 65 Theme 1: Making sense of the illness Participants described the myriad of ways in which they endeavoured to make sense of their diagnosis of and life with IBD. In particular they mentioned ways they experienced internal conflict over the cause or trigger factors of their illness and subsequent periods of particular ill health, a lack of understanding and knowledge as to what their diagnosis entailed and the misperceptions of others as to what the participants were experiencing and the nature of IBD. Cause and conflict. Participants portrayed their difficulty in dealing with the ambiguity of their illness genesis or exacerbation in a number of ways, with some suggesting an inner conflict that perhaps the development of their illness was of their own doing or preventable. For example, one participant said, "You just want answers and a lot of people ask well why I've got it and nobody (medical professionals) can ever tell you why you've got Crohn's disease, if it's your lifestyle, your diet, it's hereditary, you don't know" (Daniel, 39, CD). Other participants had their own theories as to what may have caused their illness. For example, Janet believed her illness precipitated as a result of stress stemming from a prior abusive relationship: "At the time I was being beaten up by my boyfriend, like a violent relationship, so I was like really, really stressed out. But I don't know, like science obviously speaks differently but I think things like that that's probably...I don't know I never had any issues before and that's like the only time I got ill and then like that's, well I think that's what triggered it off anyway" (Janet, 29, CD). 66 Lack of understanding. A sizeable number of the participants also described how they were oblivious or dismissive as to what their diagnosis meant in terms of its potential severity and how the disease may impact their future. Phrases such as "I didn't know anything about it" and "I wasn't educated in it that well" were common. For example, one participant admitted: "I suppose in the early stages I thought it was a bit of a oh nothing to bother about, no worries and all that lot. Nothing really to be concerned about. To now I look at it as a serious, serious illness, ya know, and it's not something to be taken lightly" (David, 48, UC). Early treatment success also seemed to lead some participants into a false sense of security as to their prognosis: "When I was initially diagnosed I didn't really think much of it which probably sounds terrible....And because I think the treatment I was put on initially worked so well and I went into remission I then just didn't really think of it" (Jackie, 29, UC). Misconceptions. The final hurdle participants faced in terms of making sense and coming to terms with their illness was dealing with the misconceptions of others as to what IBD entailed for the participant. The majority of comments were received from family members. One participant described her disappointment at her initial lack of family support for their downplaying of the severity of her diagnosis: "When I first came out of hospital the last time my nan just called it a dicky tummy and I had IBS, so I felt pretty upset about that because that made me feel really like they didn't understand what I went through, and they should've because they're my family" (Andrea, 21, UC). 67 Meanwhile, a large number of participants simply stated a lack of public awareness in relation to IBD and the difficulty in living with an 'invisible illness' that isn't instantly apparent to others: "It's only when you tell people that you've got Crohn's disease and the fact that nobody had a clue, oh what's that then, you know what I mean, that's the sort of response you get, and nobody actually thinks there's anything wrong with you, and it's one of those invisible illnesses that you look OK on the outside but on the inside is a totally different story" (Daniel, 39, CD). Participants therefore described a multitude of ways in which making sense of their diagnosis of IBD had been challenging. Experiences regarding understanding the cause of their diagnosis and health deterioration, to understanding the capabilities of IBD in terms of its ability to impinge on future health and prospects and having to deal with the lack of cognizance and sometimes insensitivity of other's perceptions of IBD were predominantly negative. The self-questioning demonstrated by those seeking to understand the cause of their diagnosis and the frustration at dealing with a lack of understanding from others elicited a consistent sense of bewilderment and segregation that seemed mostly homogenous throughout the accounts. Theme 2: Impact of IBD All participants regardless of disease severity described how IBD had impacted their lives on multiple fronts, encompassing a plethora of complex physical, social and psychological obstacles. Physical. With regards to the physical challenges of IBD, a number of the participants spoke generally of the adversity of having surgery and experiencing sometimes numerous 68 hospitalisations. The challenge of overcoming the physicality of surgery was apparent in one participant's account who stressed the "physical toll" it had taken: "The first surgery, ya know, it's a massive thing and it's a big, physical toll on your body and, ya know, the recovery period is quite hard. It's like a hard recovery to go through" (Jackie, 29, UC). A number of the interviewees also described their struggle with combating the fatigue that is predominant amongst IBD sufferers. However, of particular note was one participant's explanation of how even in remission their struggle with fatigue was ongoing, "I mean I know that I'd rather be tired and in remission than be flaring, it just seems strange that the fatigue has been more of an issue when I've been in remission" (Diane, 28, UC). Social. With respect to the social issues faced by the interviewees, these too were multi-faceted. One area it affected in particular was the participants' ability to maintain relationships with their partners, with one participant describing how it was effecting their intimacy, "I've got to prioritise what I spend my energy on and at the moment I'm spending my energy on my job rather than my partner. And most of the time he's fine about that, but occasionally it can cause an argument" (Diane, 28, UC) Further to this, participants almost unanimously expressed a difficulty in keeping up with social events, finding it difficult to commit due to the fear of potentially embarrassing symptoms in public. One participant mentioned, "I used to enjoy going out for a beer and all that with the lads and stuff like that, but that sort of put me off going out because of the, you know the situation you find yourself in 69 wanting to find a toilet if you need to go to the toilet, and you know just going into the unknown" (Daniel, 39, CD). However, despite the illness putting a strain on these relationships, one participant voiced the opinion that was shared by a number of the interviewees in that their illness strengthened the bonds with their closest friends, "I think if anything those friends have become closer because I think they feel I've shared a lot more with them now because they know so much about it and they know how hard it's been, they've been there in the hospital and wherever else and they're still with me when I feel terrible so they see it face to face" (Amy, 30, CD). Psychological. With regards to the psychological impact of IBD, the majority of participants complained of a common dilemma in dealing with a compromised self-identity. Specifically, the interviewees expressed grievances of being treated differently to others. This desire to be treated normally was made clear by one participant who said, "I don't want people to adapt how they are with me just because of my illness, I'd prefer people just to sort of treat me as they always have" (Elizabeth, 31, CD) One participant explained how her struggle to avoid being consumed by her illness was ongoing, "I still find it hard to talk about how I feel and I struggle to find a fine line between constantly complaining or constantly talking about it and not talking about it at all and I still don't think I can get there quite yet" (Amy, 30, CD) However, some interviewees had seemingly managed to distance themselves from their illness label, one participant doing so through the help of a counsellor: 70 "It made me feel like it's not colitis and Andrea , it's Andrea with colitis, ya know, I was always putting it like at the front of my mind when I shouldn't have been, I should have been thinking of myself more" (Andrea, 21, UC) Participants therefore described how the impact of living with IBD had been challenging in three distinct domains. Physically, participants reported the struggles involved with surgeries and hospitalisations, as well as the persistent battle with fatigue. Socially, their concerns ranged from issues surrounding intimacy with partners to the wider problem of socialising with friends. However, some found their diagnosis brought them closer to friends and significant others as they became familiar with the illness. Finally, some participants highlighted a psychological battle whereby the struggle to maintain their self-identity and not see themselves or be seen by others as an ill person was evident. While some were able to manage this dilemma, for others it was clearly a struggle. Theme 3: Feelings of IBD It was apparent that there were three dominant emotions that encapsulated the IBD experience. Anxiety. This was mainly displayed in the context of the initial fear and worry that the majority of participants alluded to in the face of their developing symptoms and upon diagnosis. This underlying angst was echoed by a large number of the participants by the relief that they expressed upon realising they were suffering from IBD rather than bowel cancer. For example, one participant said, "I remember at the time thinking this is really weird I'm so relieved, and I was crying and I said to him I'm not crying because I'm upset, I'm crying because I'm just so relieved that 71 you haven't told me I've got cancer...the fact that it's a chronic illness didn't really sink in I guess for a few months" (Gemma, 41, UC). Being overwhelmed. Specifically, this notion describes the helplessness that many of the participants displayed. The interviewees explained how the chronicity of their illness was an aspect of living with IBD that was difficult to handle: "Sometimes I think it can get you a little bit down because you know you've got this disease forever and nothing's gonna change, no cures" (Elizabeth, 31, CD). For others this helplessness was more pronounced, with one participant even mentioning feeling suicidal during a hospitalisation that had prevented them from attending interviews for a new job: "I was getting a bit tearful and I was actually thinking about the most convenient way to die even though I'd been through worse probably in previous years" (John, 30, CD). Animosity. Finally, participants described the animosity they felt towards their illness, characterised by feelings of resentment and frustration. One participant expressed their resentment towards their Crohn's Disease in its entirety, "In terms of attitude towards Crohn's I just hated it, I hated everything about it, I didn't want to talk about it, I resented it and it was just a very negative attitude towards it" (Daniel, 39, CD). Meanwhile, one participant expressed their resentment in the form of their jealousy of others who were not in their predicament, "There's been times in the past when I've felt angry about it, I felt 'why me', when I've just felt jealous of anyone who doesn't have to take medication every day" (Diane, 28, UC). 72 However, despite this overarching sense of animosity towards experiences of living with IBD, there were some participants who did state that they were happy despite their circumstances, as described by one patient: " I'm happy it's happened and definitely in a weird way happier than I was...I don't know if I was as happy as I was before I was diagnosed but I'm definitely happier than I have been" (Jason, 25, CD). Participants therefore described how there were a number of negative feelings towards their illness. However, despite the resentment that participants displayed towards numerous aspects of life with IBD, there was a resounding acknowledgement that positivity was essential, with some even stating they were happier than they had ever been since their diagnosis. Research has supported the existence and prevalence of depression (Graff et al., 2009) in the IBD population and while the current study supports this to an extent, these findings also suggest that positivity and happiness are possible. Overarching theme: Uncertainty Transcending these aforementioned themes was a sense of uncertainty which was particularly evident when the participants discussed the possible side effects and efficacy of treatment, apprehension associated with having surgery and the possibility of relapsing. Side effects. Participants regularly expressed their uncertainty with regards to the possible side effects of their medications. The majority expressed general concern and trepidation over the cumulative effects that their “toxic” medications could have: “I think that's my biggest problem I have is all the medication, and how toxic they are. And the side effects that come with medication and you have to take another drug to counteract the side effects and there's always in the back of my mind” (Megan, 32, UC). 73 The resounding majority of the participants’ concerns though surrounded their relationship with steroidal treatment, with many complaining of a cycle of remission and relapse when on steroids: “Had the first flare when I was diagnosed, was on steroids, came off steroids and flared again, and basically the first year after diagnosis was pretty much sort of flare, steroids, flare, not really remission, so that was pretty tough in terms of thinking well am I ever gonna get better” (Chloe, 33, UC) Surgery. Another source of uncertainty was apparent when some of the participants discussed their thoughts on surgery. Some of the interviewees described how numerous surgeries had caused them to question whether it was worth it: “I've had lots of negativity with it, surrounding it, obviously I've had lots of surgery and after each surgery not working you kind of lose a bit of hope that it's gonna ever get better” (Katie, 28, CD). This also rang true for another participant despite admitting they were now “much healthier”: “For me this surgery was something that even though I did it I still sometimes think oh I wish I hadn't done it. Even though now I feel so much healthier” (Jackie, 29, UC). Relapse. Finally, uncertainty was evident when participants expressed foreboding over the possibility of relapsing. For the majority, the prospect of relapsing was associated with the potential for loss: “I get really, really worried about it because when I had that massive flare I literally felt like I lost everything, like I lost my job and I had to drop out of college and I had to stop 74 doing all my drama classes and stuff and literally it just felt like the end of the world” (Rebecca, 19, UC) The revelation by one participant who admitted not feeling “fully adjusted” was particularly striking, that even though they were aware of their rumination it was difficult to overcome due to the fluctuating nature of their illness, “I don't think I'm fully adjusted because I think you could have a flare that's just totally different, you could have one that just knocks you for six and you're ill for a year or something, so I feel pretty adjusted but I sort of feel conscious that I don't wanna feel quite like that because that could all get knocked away” (Chloe, 33, UC) The uncertainty associated with medication and surgery compounded with the ambiguity surrounding the recipe for remission, and the fact that some of those who achieved remission felt uncertain of how long it would remain, sheds light on the difficulty of dealing with this relapsing-remitting disease. Particularly interesting too was the fact that the most feared repercussion of relapse was that of loss rather than pain for example. Overall, this uncertainty among the sample portrayed IBD as an illness that is still challenging even during remission. Final theme: The new normal This previous theme of uncertainty was counterbalanced by our final theme of the new normal. Participants displayed key psychological, social and behavioural methods of coping by which they attempted to cope with their illness and overcome its uncertainty. For example, some showed acceptance which involved a sense of letting go and accepting there may be obstacles in the future regarding their health but that they would be manageable: 75 " I'm happy, I've accepted this is what I've got and this is how it's gonna be so it's generally fine... I was scared and upset for a long while, but as I say that comes and goes, it's still gonna happen occasionally" (Amy, 30, CD) Some participants also alluded to the need to be resilient in dealing with IBD, using phrases such as “I refuse to let it beat me”, “just got to get on with it”. One participant discussed how he had to remain resolute in the face of his colectomy procedure to remove his large intestine, “I remember when they said they were going to remove it and I sort of, I thought you can either be upset about it and down or you can get on with it" (Ian, 36, UC) Participants also spoke of the importance of developing relationships that engendered a feeling of a shared experience, "I'm a member of a few forums and you discuss things with other people and you read what other people's problems are and you realise you're not alone" (Malcolm, 44, UC) Participants also almost unanimously spoke of the importance of endeavouring to live a normal life: "I have to still take child my out, still have to take him to activities and carry on as normal" (Emily, 32, UC) By employing these coping techniques, patients with IBD attempt to resolve the uncertainty of their illness and combat, for example, the misconceptions of others as to what their illness means, and achieve an equilibrium that balances their previous identity of a 'normal person' with their new identity of a person with IBD living a life that is as normal as possible. Consequently, it is posed that the ultimate goal of living with IBD is to achieve a 'new normal'. Indeed, participants alluded to the understanding that a normal life is possible 76 but that perhaps it would not be the normal they had once known. In order to reach this 'new normal', it seems that one's identity as a patient with IBD must be counterbalanced by encouraging one's sense of self and identity as a person who manages a chronic illness as a part of their life rather than a patient whose existence revolves around this illness. This supposition is encapsulated by two quotes in which both participants suggest an acceptance of uncertainty and that regardless of the fluctuations in their illness they are living the life they now have and are determined to live it to the fullest: " I think it takes a long time to work out...well to just allow yourself to try and think about your life beyond having Crohn's so when I've kind of sat down and thought about well I can still have a full life regardless, I can still do the things I want to do it's just maybe gonna be a little bit harder and it's a big revelation" (Amy, 30, CD) "I live every day as it comes...as it gets thrown at me. There's good days and bad days. I always try and set myself goals throughout so that I've got, however small, something to work towards, because I think if you get too wound up about what's happening or what's happened then you don't move forward. But it's like even after transplant there's bad times, ya know, and it can happen out the blue still but you've got to enjoy the good times so that's all I tend to do really. I don't see my life as any other person" (Katie, 28, CD) 2.5 Discussion 2.5.1 Summary of findings The present study explored how people with IBD experience their condition and highlighted three core themes relating to 'making sense of the illness', 'impact' and 'feelings'. Transcending these three themes was an overarching notion of uncertainty which in turn was resolved through a process of coping, resulting in a sense of 'the new normal'. In particular, 77 participants described utilising a number of coping mechanisms to enable them to counterbalance their identity as a patient with IBD with their identity as a person who manages a chronic illness as part of their life, rather than a patient whose existence revolves around their illness. 2.5.2 Links to literature This concept of the new normal finds reflection in notions such as that of resolution, adaptation and adjustment which is supported by, for example, the work of Charmaz (1983) who argued that patients with chronic illnesses experience a "loss of self", as was demonstrated in the current study. Patients however have agency in reconstructing their identities in the midst of illness and are thus able to redefine what it means to be, in this case, a patient with IBD but also the person who they were before diagnosis. As patients make sense of their illness and derive meaning through interaction with other patients they may develop a new understanding of their illness (Blumer, 1969) and may alter their sense of self from a person living an abnormal life due to having a chronic illness, to one that is living a new type of normal life with a chronic illness. It is also supported by Breakwell's (1986) 'identity process theory' where it was argued identity is dynamic and is altered through a process of assimilation-accommodation. In this process the person assimilates new personal information such as values or attitudes as well as social information through interpersonal networks which adjust the existing identity structure, supporting once again the notion of an identity change in the current IBD sample from abnormal to a new normal. Finally, our hypothesis relates to 'cognitive consistency theory' since it argues that individuals are motivated to act in ways and have thoughts, values and beliefs that are consistent with their sense of self (Stryker, 1980). When one is unable to achieve this consistency then cognitive dissonance occurs which motivates the adoption of behavioural and/or cognitive changes in order to realign one's situations, behaviours, attitude and beliefs back in line with their 78 aforementioned sense of self. This is true even if it requires the loss of a previously valued identity. This relates to the realisation of a number of the interviewees that they were unable to cope physically in their current state compared to their life before IBD. As such, many accepted this fact with numerous participants reporting taking on a decreased work load and lowering the expectations and demands they put on themselves. This altered sense of self, in our case the 'new normal', may lower the cognitive dissonance the patients experience as there is less failure experienced when comparing to their previous notion of 'normal'. 2.5.3 In summary Overall, the current study attempts to conceptualise adaptation where previous literature has not fully done so. Verissimo (2008), for example, mentioned how the Inflammatory Bowel Disease Questionnaire, used for measuring health related quality of life in IBD, is a construct that contributes to a better understanding of a patient's adaptation, but like much of the IBD literature it is not solely focused on attempting to conceptualise adaptation, rather it provides us with a measure by which we can associate one's level of adaptation. We can now apply this knowledge of a 'new normal' into developing a measure whereby one may quantify a patient's degree of adaptation to their illness which may have therapeutic applications. As such, the results from the current study will be used to form the basis of a new measurement of adaptation that could be used to explore the role of adaptation on psychological wellbeing, such as quality of life and mood, as well as physical wellbeing, such as disease severity, progression and recovery. 2.5.4 Methodological limitations The study is not without its limitations however. It is arguable that our deductions are based on a specific subset of patients with IBD due to our recruitment method. As participants were all part of online support groups it is possible that certain aspects of 79 adaptation that were raised, such as the importance of engendering a sense of shared experience through social networking, are not representative of patients with IBD who do not seek online support, thus bringing into question whether our notion of adaptation is transferable to all IBD patients. In conjunction with this, all participants were from the UK, again raising questions over the external validity of the findings. While the decision to recruit participants exclusively from the UK was done for reasons of pragmatism, it is arguable that adaptation as it has been conceptualised from the study findings are again not representative of all patients with IBD, with patients from other countries and cultures possibly experiencing the process of adaptation differently. Also a potential limitation of the study is that the disease activity of the participants was not accounted or controlled for. While some research has shown disease activity to be an important factor in the psychological functioning of patients with IBD (Vidal et al., 2008), there is much research that also supports the notion that neither the coping strategies (Larsson et al., 2008), nor the QoL (Magalhaes et al., 2014) of patients with IBD are altered by disease activity. Similarly, Drossman et al.'s (1989) research stated previously suggests that patients are perhaps more concerned with functional status and disease concerns than disease activity, further justifying the decision to recruit participants regardless of the current severity of their disease. With regards to the framework used for the data analysis, while thematic analysis was deemed an appropriate paradigm by which to analyse the data from the current study due to its robustness in terms of being both flexible but also capable of providing a complex account of the data, there are some limitations in taking this approach. One of these limitations is that in using an analysis tool that is so flexible, the associated drawback of this is that the range of conclusions that can be drawn from the data are broad, as indeed Braun and Clarke (2006) themselves admitted. As such, the conceptualisation of adaptation that has been put forward by the current study is undoubtedly subjective to a degree. 80 2.5.5 Conclusion To conclude, patients’ accounts of living with IBD centred around the ways in which they made sense of their condition, its impact on their lives and the feelings it generated. Permeating these experiences was an ongoing sense of uncertainty which drove patients to employ a range of behavioural, social and psychological coping methods in order to achieve a sense of a ‘new normal’ by finding an equilibrium between their lives before and after diagnosis. These findings will now be used to inform a new measure of adaptation to IBD which is the focus of Chapter 3. 81 Chapter Three Study 2: The development of a new measure to assess adaptation in patients with IBD and its utility in predicting QoL: a cross-sectional study 3.1 Overview The previous chapter utilised a qualitative study to explore and conceptualise the notion of adaptation in patients with IBD. This was achieved through conducting semistructured interviews with patients, exploring the ways in which these patients had responded emotionally, cognitively, socially and behaviourally in response to their illness from the time of their diagnosis up until the time of being interviewed. Adaptation in this context was subsequently concluded as being achieved by finding an equilibrium between one's life before and after being diagnosed, finding a sense of a 'new normal' in the process. The current chapter will now build upon this prior qualitative study in order to develop a new measure of adaptation. The results of the qualitative study will guide the formulation of this novel measure to assess adaptation to IBD which will then be refined psychometrically. This measure of adaptation will then be associated with measures of both sense making and QoL to assess whether the construct is synonymous with either construct or measuring something unique. This chapter will then also assess the ability of this new measure to predict outcomes in those with IBD in terms of QoL and its ability to predict this outcome over and above the predictive ability of a measure of illness representations. 82 3.2 Introduction 3.2.1 Literature review IBD is an all-encompassing problem for patients, challenging them on a multitude of levels contributing to psychological, social and behavioural problems and resulting in poor quality of life. In line with this, Sajadinejad et al (2012) concluded their review of the evidence by emphasising the need for good adaptation in order for patients to deal with the symptoms of IBD and its psychological impact. To date, however, little is known concerning what good adaptation means to patients with IBD or how it can be measured. The aim of the present study is to operationalise adaptation as a means to develop a new measure to evaluate adaptation in those with IBD. Although little has been written about adaptation in patients with IBD, much chronic illness research has investigated adaptation (and related concepts including adjustment and acceptance) in patients with a multitude of different health backgrounds. For example drawing upon Cognitive Adaptation Theory (Taylor, 1983), studies have explored adaptation in those treated for coronary artery disease (Helgeson, 2003), late-stage cancer of varying types (Christianson et al., 2013), skin cancer (Czajkowska et al., 2013) and type 1 diabetes (Helgeson et al., 2014). This approach is framed by three underlying processes: a search for meaning, attempting to regain mastery and self enhancement. In contrast, other studies have drawn upon the Self Regulatory Model (Leventhal et al, 1980) focusing on conditions such as Chronic Fatigue Syndrome, eating disorders and arthritis (Moss-Morris et al., 1996; Marcos et al., 2007; Pimm & Weinman, 1998). From this perspective adaptation is seen as a product of illness representations, taking a limited approach to the lived illness experience and thus neglects the biopsychosocial nature of the condition. Furthermore, the operationalisation of the notion of adaptation remains unclear. 83 As a means to capture this broader approach to IBD, Devlen et al.(2014) developed their own conceptual model of the impact of IBD which demonstrates the interconnectedness of patients’ bowel and systemic symptoms not only with the psychological impact of the disease, but also with the impacts of treatment, relationship difficulties, social and leisure activity and work, school and parenting limitations. However, Devlen et al (2014) also neglect to operationalise adaptation leaving it as the product of a number of illness consequences rather than an entity in its own right. Likewise, whilst Voth and Sirois (2009) measured adjustment to IBD, they operationalised it as a composite of coping efficacy, acceptance and helplessness rather than as a standalone construct. A similar approach can also be seen in other research on IBD which has emphasised adaptation as a form of coping (Moskovitz et al., 2000; Jones et al., 2006; McCombie et al., 2015). Therefore, whilst models such as the CAT or SRM could be used a basis to study adaptation in IBD their focus on sense making neglects the broader impact of this condition and leaves the definition of adaptation unclear. Furthermore, research specific to IBD may be broader in its perspective, but again either offers an unclear definition of what this construct means or simply equates it with other psychological processes. In line with the conclusions drawn by Sajadinejad et al (2012), however, the present paper argues that adaptation is core to the experiences of those with IBD. In addition, it also argues that in order to be measured effectively this construct requires both clear conceptualisation and operationalisation. This was the focus of a recent qualitative study which explored how patients adapted to their IBD as a means to develop the measure presented in the current paper (see Study 1 - Matini & Ogden, 2015). For this study, 20 patients were involved in semi-structured interviews concerning their experiences of adapting to IBD. Questions revolved around how patients had changed, if at all, from the time they were diagnosed until the time of interview with regards to how they felt, thought 84 and behaved with respect to their illness. The results were analysed using thematic analysis and indicated that adaptation involved the search for a ‘new normal’ with patients striving to find psychological, emotional and behavioural equilibrium between their identity as a patient with IBD and that of being a person existing beyond their illness identity. This emphasis on sustaining equilibrium reflects Moss-Morris’ (2013) recent working model of adjusting to chronic conditions which built upon more generic theories such as CAT and the SRM to highlight how patient behaviours and the chronicity of an illness are crucial aspects in enhancing understanding of adaptation. However, the results from the qualitative study by Matini and Ogden (2015) also emphasised the relational processes that occur between the patient and their illness. Moss-Morris (2013) does state that if a disease becomes progressive, factors such as acceptance and self-compassion may become more important, touching on the nature of adaptation as an iterative, ongoing process. What is missing, however, is the idea that the patient and the illness are not two separate entities, a notion highlighted by Matini and Ogden (2015) who concluded that patients with IBD demonstrated an identity that lay somewhere along a spectrum from an illness identity to a person identity. Accordingly, the self and the disease are better conceptualised as interconnected and in a dynamic dyad, rather than the disease being a separate entity that the patient simply appraises. This parallels Breakwell’s (1986) identity process theory which argued that identity is dynamic and is altered through a process of assimilation– accommodation whereby a person assimilates new personal information through interpersonal networks which adjusts the existing identity structure. This finds reflection in evidence indicating that cancer survivors experience more positive adjustment outcomes and psychological wellbeing after adopting a ‘survivor identity’ (Chambers et al., 2012; Park et al., 2009). To date, however, this has not been explored in the context of IBD. 85 In summary, it is clear that the notion of adaptation is central to the experiences of those with IBD and although more generic models such as CAT and the SRM provide some insights into this construct, it remains poorly conceptualised and therefore difficult to measure. Furthermore, whilst the working model (Moss-Morris, 2013) highlights the key role of equilibrium, adaptation again remains unclear. Therefore the aim of the present study was, firstly, to draw upon the findings of a recent qualitative study as a means to conceptualise adaptation as involving the striving for psychological, emotional and behavioural equilibrium which involves a ‘new normal’ reflected in a balance between the identity as a person and that as a patient with a disease. Furthermore, the study aims to develop a new measure of adaptation to IBD based upon this conceptualisation which could be used to quantify adaptation in this patient group. Finally, the current study aims to determine whether the adaptation to IBD questionnaire measures something distinct from sense making, in this case with regards to illness representations, and QoL; to investigate whether the adaptation to IBD questionnaire is useful in predicting QoL in patients with IBD; and lastly to assess whether the process of adaptation, as measured by the questionnaire, aids in the prediction of QoL scores over and above measures of sense making alone. QoL in the current study will be measured differently for those patients with IBD who have a stoma and those without a stoma since the IBDQ uses slightly different items to account for differences in the lived experience between the two groups. The results will therefore discuss how well the adaptation to IBD questionnaire subscales predict QoL scores for those with a stoma (using the IBDQ-S) and without a stoma (using the IBDQ) separately. 3.2.2. Steps involved in development and validation of the adaptation to IBD questionnaire For clarity, Table 2 below outlines the scope of the current study in terms of what steps of a scale development and validation will be undertaken and what steps have been omitted. 86 Table 2: Scale development and validation steps included and not included Steps included Steps not included Item generation Confirmatory factor analysis Data screening Test-retest reliability Exploratory factor analysis Content validity Reliability assessment Face validity 87 3.3 Method 3.3.1 Design The study used a quantitative, cross sectional design. 3.3.2 Sample Participants were recruited as part of an opportunity sample from social networking sites (Twitter and Facebook), as well as dedicated IBD online support website forums. Participants were required to be at least 16 years of age, capable of providing informed consent, self-reporting a diagnosis of IBD and able to understand English. Participants described their gender, age, highest level of education, ethnicity and IBD diagnosis. In total 388 participants began the study and of these 309 completed the study giving a completion rate of 80%. Of the 309 completers, seven were diagnosed with ‘indeterminate colitis’, 83 with UC and 217 with CD. Two participants were excluded from the analysis as their diagnosis was described as IBS. Of the remaining 307 participants, 52 were male (16.9%) and 255 were female (83.1%) and the mean age was 34.14 years for the sample (range = 1668 years). The most common level of qualification for the sample was undergraduate degree (n=122) and the most common ethnicity was White British (n=154). Of the 307 participants, 41 reported having a stoma, while the remaining 266 participants reported not having a stoma. 3.3.3 Procedure The study was advertised on social networking sites and support groups providing brief details concerning the aims of the study, a contact for those requiring further details and a hyperlink to the Qualtrics study page with the online adaptation to IBD questionnaire (See Appendix I), the Brief Illness Perception Questionnaire (See Appendix J), the Inflammatory 88 Bowel Disease Questionnaire (See Appendix K), and which also included an information sheet (See Appendix F), consent form (Appendix G) and debriefing page at the end for those taking part (Appendix H). Ethical approval was obtained from the University Ethics Committee (See Appendix N). 3.3.4 Measures i) Adaptation to Inflammatory Bowel Disease questionnaire: Participants completed the original, 40-item version of the Adaptation to Inflammatory to Inflammatory Bowel Disease questionnaire (A-IBD; See Appendix I). For this study, QoL will be predicted based on the final version of the A-IBD that will be formulated in this study. This measure assesses the process of adaptation to IBD based on one's degree of 'patient identity', 'person identity', 'acceptance' and 'expectations'. ii) Brief Illness Perception Questionnaire: Participants completed the Brief IPQ (Broadbent et al., 2006; See Appendix J) which assesses the process of sense making in terms of cognitive and emotional representations of illness relating to consequences, timeline, personal control, treatment control, identity, concern, coherence and emotion. Permission to reproduce this measure has been granted by Elizabeth Broadbent. iii) Inflammatory Bowel Disease Questionnaire: The outcome measure of interest involved in this study is the IBDQ (Guyatt et al., 1989; See Appendix K). This measure of subjective health demonstrates excellent validity and reliability (Pallis et al., 2004) as a tool to measure health-related quality of life in adult patients with IBD and has been used extensively in research and clinical trials since 1997 and was thus deemed as the most suitable way by which to measure HRQoL in this sample. Within the questionnaire are 32 questions, each with a graded response numbered 1 through 7 usually indicating a response ranging from "all of the time" to "none of the time". The questions relate to four categories of HRQoL in IBD, 89 namely 'bowel systems', involving 10 questions such as "How frequent have your bowel movements been during the last two weeks?", 'emotion health', involving 12 questions such as "How often during the last 2 weeks have you felt frustrated, impatient, or restless?", 'systemic systems', involving five questions such as "How often has the feeling of fatigue or of being tired and worn out been a problem for you during the last 2 weeks?", and 'social function', involving a further five questions such as "How often during the last 2 weeks have you been unable to attend school or do your work because of your bowel problem?". All 32 questions were phrased with reference to the last 2 weeks of the participants' experiences. 266 participants completed the IBDQ. There is an alternate version of the questionnaire, the IBDQ-S (See Appendix L), designed for patients with IBD with an ileostomy or colostomy appliance. This form of the questionnaire varies from the standard IBDQ only slightly on six questions in order to phrase the items in a more appropriate manner for patients of this type, replacing questions such as "How much of the time during the last 2 weeks have you felt embarrassed as a result of your bowel problem?" with questions such as "How frequently have you had to empty your colostomy or ileostomy appliance during the last two weeks?" 41 participants completed the IBDQ-S. Permission to reproduce this measure has been granted by McMaster University. 3.4 Results Developing the new measure of adaptation to IBD (A-IBD) 3.4.1 Conceptualisation The previous qualitative study by Matini and Ogden (2015) identified three core themes from the experiences of these adapting to IBD. These were making sense of the illness, impact of IBD and feelings of IBD. These find close parallels to models of sense making and illness representations in chronic illness (Taylor et al, 1983; Leventhal et al, 90 1980). It was argued, however, that transcending these themes was the key theme of uncertainty which was managed through a process of striving to find a new normal. Central to this process was the balance between the identity as a person (without the illness) and the identity as a patient (with the illness). Furthermore it was argued that this balance was achieved via a degree of acceptance and expectations of a positive future. In line with this, the present study conceptualised adaptation to IBD in terms of four constructs: identity as a person; identity as a patient; acceptance; expectations. This perspective draws upon the working model by Moss-Morris (2013) but also reflects theories of identity (Breakwell, 1986). 3.4.2 Operationalisation The four constructs outlined above were each operationalised using 10 items derived from the qualitative results from Matini and Ogden (2015). The items were intended to reflect as closely as possible the most pertinent aspects of the aforementioned study’s results, mainly the notion of ‘person identity’ and ‘patient identity’, but also the underlying themes of ‘acceptance’ and ‘expectations’. Examples are as follows: Identity as person: "I exert myself just as much as I used to before my diagnosis"; "I go out and socialise regardless of any symptoms I may be experiencing"; "I try and live a normal life like everyone else" Identity as a patient: "I fear that my illness will stop me achieving my goals in life"; "If I exercise then my symptoms will return"; "Life will never be the same again" Acceptance: "Some days I will not be as productive as I want to be"; "I am comfortable discussing my symptoms with my family and close friends"; "I feel like my illness is a burden" 91 Expectations: "I will be cured from my illness one day"; "I will live the life I had before being diagnosed"; "I will achieve everything I always wanted to in life" This created the 40 items included in the initial questionnaire which were each rated on a 5 point Likert scales ranging from 'strongly disagree' (1) to 'strongly agree' (5). These response options were chosen since they enabled endorsement of a statement. Enabling participants to endorse statements by agreeing or disagreeing encourages honesty and reflection as participants align themselves with other people in a similar situation. Higher scores for each item reflect higher scores on each of the four subscales. 40 initial items were included to enable items to be excluded throughout the process of questionnaire development. 3.4.3 Assessment of the scale’s psychometric properties The psychometric properties of scales were assessed and refined using: i) data screening; ii) the principle axis factoring method of factor extraction to reduce the items into a factor structure that was both statistically and semantically coherent; iii) reliability assessment using Cronbach's alpha. Data screening: Using a range of criteria based upon the ratio between sample size and number of items (eg. Comrey, 1973; Kass & Tinsley, 1979; Field, 2005) the sample of the current study (n=307) was deemed appropriate for a factor analysis of 40 items. Furthermore, the Kaiser-Meyer-Olkin (KMO) statistic was 0.880 indicating that the sampling adequacy of the current data set was ‘meritorious’. To explore inter correlation between variables a correlation matrix was computed for all 40 items. From this, Bartlett’s test of sphericity statistic was highly significant (p<.001) indicating that the data was factorizable. However, the correlation matrix was further assessed to investigate whether any one variable failed to 92 correlate with at least one other variable at a value of less than 0.3 (Field, 2005). As such, 4 items (“I worry that when I go out with my friends I may suddenly need the toilet”, “I spend time researching medications or other treatment options”, “I am comfortable discussing my symptoms with my family and close friends” and “Surgery will completely turn things around for me”) were excluded from the analysis. Finally, the degree of multicollinearity was assessed using the determinant statistic (Field, 2005). The determinant was 0.00000105 which is less than the desired value. However, no two items were correlated above r = 0.8 suggesting that factor analysis was an acceptable approach. Principle axis factoring: Principle axis factoring was chosen as this was an exploratory study. This method takes into account both the latent variables that contribute to the underlying factor structure and the difference between shared and unique variance and, as the data was largely not normally distributed (Fabrigar et al., 1999). An oblique rotation method was chosen for the current analysis to aid interpretability and as it allows for the underlying factors to correlate with one another (Field, 2005). The data was then run through a series of factor analyses as follows. Criteria for inclusion of any given item were: a cut off of factor loading >0.4 (shown in bold in the factor loadings tables below), a communality statistic of >0.3 and no cross loading. Solution 1: Initial principle axis factoring revealed 11 factors that had eigenvalues greater than 1.0 and which explained 21.3%, 4.9%, 4.2%, 4.0%, 2.4%, 1.9%, 1.6%, 1.5%, 1.3%, 1.2% and 1.1% of the total variance accounting for 45.3% of the total variance explained. However, the scree plot indicated a four factor solution according to Cattell (1966) Solution 2: A forced 4 factor solution resulted in factors explaining 22.7%, 4.7%, 4.2% and 3.4% of the total variance, accounting for a total of 35.1% of the total variance. The scree plot still showed a 4 factor solution. However, due to loading on more than one factor or having a communality statistic <0.3, 12 items were removed. 93 Solution 3: A forced 4 factor solution was re-run with the remaining 28 items. The scree plot again supported a four factor solution with the four factors now explaining 23.6%, 6.5%, 5.3% and 4.0% of the total variance, accounting for an improved total of 39.4% of the total variance. A further 2 items were removed due to their low communality statistic (0.152) and high cross-loading on another factor. Solution 4: The next analysis was run on the remaining 26 items. The factors this time explained 23.8%, 6.7%, 5.4% and 4.2% of the total variance, accounting for an improved total of 40.2% of the total variance explained. From this analysis a final 8 items were removed due to low communality and high cross loading. The final solution: The final solution with 18 items consisted of four factors with no cross loading and with all factor loadings>0.4. There were labelled as follows: Factor 1 was labelled ‘Patient identity’ and consisted of 6 items (eg.(“I fear that my illness will stop me achieving my goals in life”; “I feel like my illness is a burden”, “Life will never be the same as it was before my diagnosis); factor 2 consisted of 5 items and was labelled ‘Person identity’ (eg. “I exert myself just as much as I used to do before my diagnosis”; “I try and live a normal life like everyone else”; “I still keep up my hobbies and interests that I had before my diagnosis); factor 3 consisted of 4 items and was labelled ‘Expectations’ (eg. “I expect my doctor will eventually find a medication that solves my problems”; “I expect things will only get easier with time”; “I believe my fatigue will sort itself out eventually”) and factor 4 consisted of 3 items and was labelled ‘Acceptance’ (eg. “I accept that I have a chronic illness which has no cure”; “I am accepting of the possibility of future flare ups of my symptoms”; “I just have to keep moving forward with my life”). These four factors were corroborated by the scree plot, with the factors accounting for 22.8%, 8.0%, 5.5% and 4.8% with the total explained variance being 41.2%. The KMO statistic for this final solution was ‘meritorious’ at .853 and the determinant value now indicated no concern for 94 multicollinearity at .009. In addition, Bartlett’s test of sphericity was once again highly significant at p<.001. The factor loadings for each factor and its items are shown in Table 3 and the means and standard deviations for each item are shown in Table 4. The factor loadings demonstrate strong loadings for teach item on their respective factors with very little cross loading suggesting the 4 factor solution was appropriate. The standard deviation statistics meanwhile suggest a generally diverse range of responses on the 18 item scale, with the three items of the ‘acceptance’ subscale demonstrating the least variance in responses of the four subscales. 95 Table 3: Factor loadings for 18 item adaptation questionnaire (n=307) Item I fear that my illness will stop me achieving my goals in life Rotated factor loadings 1. Patient 2. Person identity identity -.16 -.63 3. Acceptance 4. Expectations .04 .05 I feel like my illness is a burden -.63 -.16 .11 .00 Life will never be the same as it was before my diagnosis -.59 -.11 -.14 -.11 I get angry or upset when I experience symptoms -.59 .13 .11 .00 I wonder about how my health will be in the future -.59 .02 -.12 .03 There is nothing I can do to make myself feel better -.52 -.03 .06 -.17 I exert myself just as much as I used to do before my diagnosis .13 .67 .14 -.06 I try and live a normal life like everyone else .00. .64 -.02 .01 I still keep up with my hobbies and interests that I had before my diagnosis .17 .63 -.02 -.00 I ignore any fatigue I may be experiencing and try to function as normal -.13 .62 -.04 .02 I go out and socialise regardless of any symptoms I may be experiencing .07 .49 -.02 .13 I accept that I have a chronic illness which has no cure -.08 -.07 -.68 .00 I am accepting of the possibility of future flare ups of my symptoms .09 -.06 -.61 -.12 I just have to keep moving forward with my life .04 .26 -.41 .08 I expect my doctor will eventually find a medication that solves my problems -.01 -.07 -.11 .68 I expect things will only get easier with time .19 .07 -.02 .59 I believe my fatigue will sort itself out eventually .09 .10 .05 .56 I will be cured from my illness one day -.10 .00 .16 .54 96 Table 4: Means and standard deviations for 18 item adaptation questionnaire Item Mean Standard Deviation I fear that my illness will stop me achieving my goals in life 3.50 1.27 I feel like my illness is a burden 3.74 1.04 Life will never be the same as it was before my diagnosis 3.93 1.09 I get angry or upset when I experience symptoms 3.88 .92 I wonder about how my health will be in the future 4.42 .74 There is nothing I can do to make myself feel better 2.49 1.05 I exert myself just as much as I used to do before my diagnosis 2.54 1.26 I try and live a normal life like everyone else 4.03 .93 I still keep up with my hobbies and interests that I had before my diagnosis 2.95 1.25 I ignore any fatigue I may be experiencing and try to function as normal 1.11 3.35 I go out and socialise regardless of any symptoms I may be experiencing 2.68 1.20 I accept that I have a chronic illness which has no cure 4.18 .82 I am accepting of the possibility of future flare ups of my symptoms 4.27 .77 I just have to keep moving forward with my life 4.31 .58 I expect my doctor will eventually find a medication that solves my problems 2.67 1.03 I expect things will only get easier with time 2.65 1.07 I believe my fatigue will sort itself out eventually 2.26 1.01 I will be cured from my illness one day 2.29 1.05 97 3.4.4 Reliability assessment Cronbach’s alphas were calculated for each subscale to assess their internal reliability. Patient identity (6 items) was 0.8; person identity (5 items) was 0.8; expectations (4 items) was 0.7 and acceptance (3 items) was 0.6. As such, the patient identity, person identity and expectations scales were found to have good internal reliability, while the acceptance subscale suggested comparatively low reliability. None of these alphas could be improved by deleting items and no items from any of the factors had a corrected item-total correlation of less than 0.41, suggesting all items correlated well with their respective factor. The 18 item scale also appeared to demonstrate sufficient face validity, highlighting psychological, emotional, social and behavioural aspects that are consistent with the notion of adaptation. 3.4.5 Final scale with response options and instructions The final, 18 item adaptation to IBD questionnaire (A-IBD) would thus be presented to prospective patients as shown below, with instructions and response options included: Adaptation to IBD Questionnaire Read the following statements and mark your level of agreement with each statement based on the response options listed below each statement. If you do not have an opinion on a particular statement, please mark “Neither agree nor disagree”. 1) I fear that my illness will stop me achieving my goals in life Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 2) I feel like my illness is a burden Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 3) Life will never be the same as it was before my diagnosis 98 Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 4) I get angry or upset when I experience symptoms Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 5) I wonder about how my health will be in the future Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 6) There is nothing I can do to make myself feel better Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 7) I exert myself just as much as I used to before my diagnosis Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 8) I try and live a normal life like everyone else Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 9) I still keep up with my hobbies and interests that I had before my diagnosis Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 10) I ignore any fatigue I may be experiencing and try to function as normal Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 11) I go out and socialise regardless of any symptoms I may be experiencing Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 12) I accept that I have a chronic illness which has no cure Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 99 13) I am accepting of the possibility of future flare ups of my symptoms Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 14) I just have to keep moving forward with my life Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 15) I expect my doctor will eventually find a medication that solves my problems Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 16) I expect things will only get easier with time Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 17) I believe my fatigue will sort itself out eventually Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 18) I will be cured from my illness one day Strongly disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree 3.4.6 Association of the A-IBD with existing measures of sense making and QoL The A-IBD sub scales were computed, descriptive statistics were run and scores were correlated with the subscales of the Brief IPQ and IBDQ. Descriptive statistics indicated that the mean 'patient identity' score was 3.66 (S.D.=0.72), the mean 'person identity' score was 3.11 (S.D.=0.83), the mean 'acceptance' score was 4.3 (S.D.=0.54) and the mean 'expectations' score was 2.47 (S.D.=0.76). The correlation matrix is shown in Table 5. 100 Table 5: Correlations between A-IBD, Brief IPQ and IBDQ/IBDQ-S Brief IPQ A-IBD Patient Cons Time IBDQ IBDQ-S P. Treat. Ident- Conc- Coh- Emot. Bowel Emot. Syst. Social Bowel Emot. Syst. Control Control ity ern erence rep sympt health sympt funct sympt health sympt .46** -.15* .34** .28** -.44** -.33** -.06 -.39** -.22** -.40** -.33** -.31** .41** -.05 -.04 Person - -.15* .34** .28** -.44** -.33** -.06 -.39** .27** .36** .37** .44** -.53** -.02 -.01 identity .48** Accept- -.07 .19** .18** .12* -.08 -.08 .33** -.10 -.00 .06 -.02 .03 -.13 .06 .05 Expect- - -.30** .26** .23** -.28** -.21** -.12* -.29** .18** .26** .27** .27** -.46** -.07 -.06 ations .25** identity ance 101 The results showed that ‘Patient identity’ was significantly positively correlated with the consequences, personal control and treatment control subscales of the Brief IPQ and negatively correlated with timeline, identity and concern. Further, ‘Patient identity’ was also positively correlated with bowel symptoms for those with a stoma but negatively correlated with all other subscales of the IBDQ. ‘Person identity’ was positively correlated with personal control and treatment control of the Brief IPQ but negatively correlated with most other subscales. However, whereas ‘Person identity’ was positively correlated with all subscales of the IBDQ, it was negatively correlated with bowel symptoms for those with a stoma. The scale reflecting ‘Acceptance’ was positively correlated with timeline, personal control, treatment control and coherence but unrelated to consequences, identity, concern and emotion or any subscales of the IBDQ or IBDQS. Finally, ‘Expectations’ was positively correlated with personal control and treatment control of the Brief IPQ but negatively correlated with consequences, timeline, identity, concern, coherence and emotion. It was also positively correlated with all subscales of the IBDQ but negatively correlated with the bowel symptoms subscales of the IBDQ. The subscales of the A-IBD therefore show some relationships with illness representations as measured by the Brief IPQ and Quality of life as measured by the IBDQ (and IBDQ-S). The highest correlation coefficient was -0.53 ('person identity' and 'bowel symptoms' on the IBDQ-S) which was moderate in strength whilst the remaining correlations demonstrated weaker or no association indicating that although this new measure shares some of the variance with existing scales it is also making a novel contribution to the concept of adaptation. It is also of note that correlations between the AIBD differed between the IBDQ and the IBDQS indicating that adaptation may have a different profile in these two populations. 102 3.4.7 Using the A-IBD subscales to predict QoL This section will begin with assessing the ability of the subscales of the A-IBD to predict QoL, as measured by the IBDQ, firstly in the non-stoma group (using the standard form of the IBDQ) and then in the stoma group (using the adjusted version to be made applicable for those with stomas, known as the IBDQ-S). Extensive data screening was carried out in both cases to ensure a sufficient regression model could be produced for both groups. The interpretation of the regression models and coefficients follows the data screening procedures. Non-stoma group Data screening – Sample size Many rules of thumb exist for recommended sample sizes in order to obtain a reliable regression model. Generally, however, 10 to 15 participants per predictor in the model is deemed suitable (Field, 2005), meaning the current analysis would require roughly 40 to 60 participants, respectively, given four predictors are involved in the current model. As such, it is apparent that the total number of participants without stomas (266) meet this suggested requirement comfortably. The non-stoma group also far exceeds Green’s (1991) more aggressive minimum recommendation formula of 104 + k where ‘k’ is the number of predictors in the model, creating a total minimum recommendation of 108 in this instance. Furthermore, the sampling size adequacy of this group was supported by the guidelines proposed by Miles and Shelvin’s (2001) graphs which illustrate the sample sizes needed when expecting small, medium and large effect sizes at different levels of power and with differing numbers of independent variables. Cohen (1988) proposes power to be at least .8, i.e. there is an 80% probability of the test correctly rejecting the null hypothesis when the alternative hypothesis is true, and based on this criteria Miles and Shevlin’s graphs suggest 103 the non-stoma group’s sample size is comfortably sufficient enough to detect a medium effect size given there are four independent variables (a minimum of 90 participants is suggested for this effect size). Data screening – Independence of residuals One assumption of multiple regressions is that for any two observations the residual terms should be uncorrelated and thus independent. In other words, they should demonstrate a lack of autocorrelation. The Durbin-Watson test assesses whether adjacent residuals are correlated with values near 2.0 suggesting no significant autocorrelation (Piegorsch & Bailer, 2005). In this instance, the Durbin-Watson statistic was 1.910 and thus sufficiently met this particular assumption. Data screening – Assumption of linearity Since the results are being generated using a linear multiple regression model it follows that the relationship being modelled must also be linear. In order to test this, a linear relationship between the dependent and independent variables collectively needed to be established and was achieved by formulating a scatterplot of the studentized residuals against the unstandardized predicted values. The plot that was generated indeed indicated a linear relationship in this instance since the residuals formed a horizontal band as opposed to showing any curves. However, a linear relationship also needed to be established between the dependent variable and each of the independent variables using partial regression plots. Here it was confirmed that all relationships between the independent and dependent variables were linear and thus met the current assumption, albeit the relationship between ‘patient identity’ and IBDQ total and ‘person identity’ and IBDQ total demonstrated a far more pronounced linear relationship than ‘hope’ and IBDQ total and ‘acceptance’ and IBDQ total. 104 Data screening – Homoscedasticity In order to check that the residuals are equal for all values of the dependent variable (in other words, the variance along the line of best fit remains similar at each point on the line), the scatterplot that was assessed with regards to the issue of linearity in the previous section (studentized residuals against the unstandardized predicted values) was once again consulted. In order to satisfy the assumption of homoscedasticity this plot needed to demonstrate an even spread of the residual values, i.e. no particular pattern should have been evident, rather there was an approximate constant spread as one moves across the predicted values axis. In the current sample, homoscedasticity did indeed seem apparent due to visual inspection of the random scattering evident in the scatterplot. Data screening – Multicollinearity Occurring when there are two or more independent variables that are too highly correlated with one another, multicollinearity causes issues in understanding exactly which of these independent variables then contributes to the variance explained in the dependent variable. Consulting first the correlations table of the multiple regression output, it can be seen that multicollinearity was not seemingly an issue since none of the independent variables correlated higher than .558, with correlations above .7 or .8 causing concern. Subtler forms of multicollinearity however are picked up by the ‘tolerance’ and ‘VIF’ statistics. Generally, tolerance values of less than .1 are a serious cause for concern and Menard (1995) even suggests values below .2 to be an issue. In this instance the lowest tolerance value was .786 and the highest was .973, thus not raising any red flags as to possible multicollinearity. The VIF is merely the reciprocal of tolerance (1 divided by tolerance) so naturally these values are also not indicative of multicollinearity. 105 Data screening – Outliers, high leverage values and highly influential points No outliers were detected for the current sample since no ‘casewise diagnostics’ table was produced in the analysis output. However, this merely detects outliers based on standardized residuals that are +/-3 standard deviations. Consulting the studentized deleted residuals in conjunction with this supported the prior finding as once again no residuals in this category were greater than +/-3 standard deviations. ‘Leverage values’ can also be consulted to distinguish if any particular observed values of the independent variable vary drastically from their predicted value as predicted by the regression model. General rules of thumb for this statistic suggest values less than .2 as safe (Hoaglin, 1992), all of which met this criterion in the current sample. Similarly, influential points can be assessed by the ‘Cook’s distance’ statistic which measures the overall influence of a case on the subsequent regression model. For this statistic, Cook and Weisberg (1982) suggest values greater than 1 may be a cause for concern, but no such values were found in the current sample. Data Screening – Normally distributed residuals It is a necessity that when running inferential statistics that the residuals (the errors in prediction) need to be normally distributed (unlike the independent variables themselves). In other words, the differences between the generated model and the observed data should be zero or negligible. A histogram of the standardized residuals with a distribution curve is one way to test this assumption. In this case, the histogram with a superimposed distribution curve appeared to show a normal distribution as was desired. In addition, the mean value was approximately 0 and the standard deviation 1, both supporting the notion that the residuals were normally distributed. Concerns for the violation of the assumption of normality of residuals was also eased by the P-P plot which showed very few deviations from normality as 106 demonstrated by most observed residuals lying along the straight, diagonal line which indicates a perfectly normal distribution. Method of regression Forced entry was the chosen regression method since no prior literature on adaptation and what constitutes it dictated substantive theoretical importance as to what independent variables we should favour as contributing more to the regression model than others. As such, stepwise, hierarchical methods were deemed not applicable. Interpreting the regression model – Total variation explained The model summary indicates a multiple regression coefficient (R) value of .671, suggesting that the linear association between the predicted scores of the regression model and the actual values of the dependent variable (quality of life as measured by the IBDQ) was moderately strong to strong. However, while this gives an indication as to the goodness of the model fit, the R2value of .450 is more indicative of this. This value accounts for how much of the variability in IBDQ scores was accounted for by the four factors, i.e. 45.0%. The adjusted R2meanwhile shows that when accounting for shrinkage that occurs if the model were applied to the IBD population (of patients without stomas) as opposed to the sample in the current study, this time a statistic of .441 was found meaning that the model would account for approximately only .9% less variance in IBDQ scores. Altogether, according to Cohen’s (1988) effect size brackets, .441 is indicative of a moderate effect size. Interpreting the regression model – Statistical significance of the model The statistical significance of the overall model was highly significant (p<.001). What this suggests is that the addition of all of the four independent variables led to a model that is statistically significantly better at predicting IBDQ scores than the mean model and is a 107 statistically significantly better fit to the data than the mean model. Altogether, this means that ‘person identity’, ‘patient identity’, ‘hope’ and ‘acceptance’ significantly predicted IBDQ scores, F(4, 261) = 53.332, p<.001. Interpreting the coefficients The ‘person identity’ and ‘patient identity’ coefficients were both statistically highly significant (p<.001), thus both were deemed significant predictors of IBDQ scores ((t(261) = 7.453, p<.001) and (t(261) = -7.338, p<.001), respectively). The ‘hope’ and ‘acceptance’variables however were not significant ((t(261) = 1.313, p=.190) and (t(261) = 1.290, p=.198), respectively) and thus did not demonstrate a convincing contribution to the prediction of IBDQ scores. With regards to the variable coefficients (B), for every increase of one point on the person identity scale, IBDQ scores raise by 17.140 (B = 17.140), while for every increase of one point on the patient identity scale, IBDQ scores fall by 19.494 (B = 19.494). Unsurprisingly, hope and acceptance were less impactful with one point on the hope scale raising IBDQ by 3.214 (B = 3.214) and one point on the acceptance scale raising IBDQ by 4.066 (B = 4.066). Stoma group Data screening – Sample size While not meeting the recommended sample size as suggested by Green’s (1991) aforementioned 104+ k formula, the current sample did meet the more liberal guidelines of Field’s (2005) 10 to 15 participants per predictor variable since there were 41 participants who completed the IBDQ-Stoma. Even more conservative suggestions have been made by Austin and Steyerberg (2015) who claim that as few as two participants per independent variable, which would amount to eight required participants in this instance,is sufficient in 108 order to estimate regression coefficients of a multiple regression model with relative bias of less than 10%. Data screening – Independence of residuals As with the non-stoma group, the Durbin-Watson statistic did not cause any concerns with regards to autocorrelation between the residual terms since the statistic was reasonably close to the previously outlined, desired value of 2 at 1.825. Data screening – Assumption of linearity The scatterplot of the stoma group’s unstandardized predicted residuals vs their studentized residuals depicted a somewhat linear relationship as the residuals appeared to be ordered in a horizontal band with no apparent curvature. The partial regression plots, while not as easily interpreted as those of the non-stoma group, did appear to satisfy the assumption of linearity, particularly in the case of the relationship between IBDQ-Stoma total scores and the ‘patient identity’ scale scores which showed a more pronounced linear relationship. Data screening – Homoscedasticity The previous scatterplot depicting the unstandardized predicted residuals against the studentized residuals of the IBDQ-Stoma group demonstrated that the residuals were evenly spread across the unstandardized predicted value axis, with no fanning or funnelling in the pattern of the data that would signal a lack of homoscedasticity. Data screening – Multicollinearity Consulting the correlations table of the regression output, although ‘patient identity’ and ‘person identity’ scores did correlate very close to the .7/.8 mark that was previously described as the point at which concerns for multicollinearity begin to arise at -.697, the 109 tolerance (and VIF) statistics allayed this fear. The lowest tolerance statistic was .588, comfortably higher than the .1/.2 mark that causes concerns for multicollinearity and since this is regarded as a stronger means of detecting this issue, this assumption was deemed to have been met satisfactorily. Data screening – Outliers, high leverage values and highly influential points No outliers were detected based on the standardized residuals (that are highlighted in the regression output should they arise). Similarly, the studentized deleted residuals brought up very little in the way of outliers besides one case where the residual was found to be greater than -3.03 standard deviations. However, since the Leverage value for this case was below .2 and Cook’s distance was below 1, this case was not deleted. In addition, in terms of Leverage values, only two cases proved to be above the ‘safe’ .2 value and in the category of ‘risky’ between .2 and .5 (.38 and .31). However, once again both cases were retained since their Cook’s distance values were comfortably below 1. In fact, all cases demonstrated Cook’s distance values comfortably below the cut off of 1, with .39 being the highest value. Data Screening – Normally distributed residuals A histogram of the standardized residuals with a superimposed distribution curve once again appeared to show a normal distribution of the residuals as desired. In conjunction, the mean and standard deviation was also once again virtually equal to 0 and 1, respectively, supporting the idea that the residuals were normally distributed. While the current P-P plot depicted slightly more deviations from normality than that of the non-stoma group, it still did not appear to demonstrate significant deviation from normality as most observed residuals lay close to the diagonal line. 110 Method of regression Forced entry was once again chosen as the regression method. Interpreting the regression model – Total variation explained The model summary indicates multiple regression coefficient (R) value of .721, suggesting that the linear association between the predicted scores of the regression model and the actual values of the dependent variable (quality of life as measured by the IBDQStoma) was strong. The R2value was .520, meaning that the for 52.0% of the variance in the IBDQ-Stoma scores could be accounted for by the four factors. The adjusted R2value was .467, meaning that if applied to the general population (IBD patients with stomas) the current model would account for approximately 5.3% less of the variance in IBDQ-Stoma scores. Altogether, .467 once again indicated this result represented a medium effect size. Interpreting the regression model – Statistical significance of the model The statistical significance of the overall model was highly significant (p<.001), suggesting that the addition of all of the four independent variables led to a model that is statistically significantly better at predicting IBDQ-Stoma scores than the mean model and is a statistically significantly better fit to the data than the mean model. Altogether, this means that ‘person identity’, ‘patient identity’, ‘hope’ and ‘acceptance’ significantly predicted IBDQ-Stoma scores, F(4, 36) = 9.766, p<.001. Interpreting the coefficients The ‘patient identity’ coefficient was statistically highly significant t(36) = -3.797, p<.05. However, the remaining three independent variables of ‘person identity’ (t(36) = .879, p = .385), ‘hope’ (t(36) = 1.163, p = .253) and ‘acceptance’ (t(36) = -.008, p = .993) were not found to provide a statistically significant contribution to the prediction of IBDQ-S scores. 111 With regards to the variable coefficients (B), for every increase of one point on the patient identity scale, IBDQ-Stoma scores fall by 25.729 (B = -25.729) for every increase of one point on the person identity scale, IBDQ-Stoma scores raise by 5.296 (B = 5.296), for every increase of one point on the expectations scale, IBDQ-Stoma scores raise by 6.631 (B = 6.631), while for every increase of one point on the acceptance scale, IBDQ-Stoma scores were found to actually fall but by a mere .067 (B = -.067) which is a reflection of its extreme lack of significance as demonstrated by its p value of .993. 3.4.8 Adaptation formula Based on the above regression models, going forward adaptation will therefore be measured both in those with and without stomas using the formula of: person identity + acceptance + expectations -patient identity. 3.4.9 Does the A-IBD predict QoL over and above the BIPQ? Non-stoma group Table 6 below displays the multiple regression analysis results when the four subscales of the A-IBD were added to the regression model predicting IBDQ scores (Model 2) compared to when the eight items of the BIPQ predicted IBDQ alone (Model 1). 112 Table 6: A-IBD and BIPQ predicting IBDQ scores R R2 Adj. R2 R2 change F Sig. 1 .760 .578 .565 .578 44.024 .0001 2 .803 .645 .628 .066 38.236 .0001 Model What these results show is that the BIPQ alone is statistically significantly better at predicting IBDQ scores than the mean model, showing a strong correlation between the two sets of scores (R=.760), that it accounts for 57.8% of the variance in IBDQ scores (R2 =.578) and demonstrates a medium effect size (Adjusted R2 =.565). When the A-IBD was added to the regression model, it was found the model was still highly significant and its four subscales predicted a further 6.6% of the variance in IBDQ scores. Stoma group Table 7 below displays the multiple regression analysis results when the four subscales of the A-IBD were added to the regression model predicting IBDQ-S scores (Model 2) compared to when the eight items of the BIPQ predicted IBDQ-S alone (Model 1). 113 Table 7: A-IBD and BIPQ predicting IBDQ-S scores R R2 Adj. R2 R2 change F Sig. 1 .771 .595 .494 .595 5.881 .0001 2 .804 .646 .494 .051 4.253 .001 Model What these results show is that the BIPQ alone again was statistically significantly better at predicting IBDQ-S scores than the mean model, showing again a strong correlation between the two sets of scores (.771), that it accounts for 59.5% of the variance in IBDQ-S scores and demonstrates once again a medium effect size. When the A-IBD was added to the regression model, it was found the model was still highly significant and its four subscales predicted a further 5.1% of the variance in IBDQ-S scores. 3.5 Discussion 3.5.1 Summary of findings Research indicates that IBD impacts upon patients’ daily lives in a multitude of ways and that adaptation to this condition is core if people are to manage their condition effectively. To date, however, although much has been written about adaptation to chronic illnesses in general, and in particular IBD, what this construct means and how it should be measured remains unclear. The aim of the present study was therefore to provide a clear conceptualisation of adaptation in the context of IBD as a means to operationalise this construct and develop a reliable measure. Conceptualisation was based upon the chronic illness literature and specifically a qualitative study of 20 IBD patients reported in Study 1 114 (Taylor et al, 1983; Leventhal et al, 1980; Moss-Morris, 2013; Matini & Ogden, 2015). From this, adaptation was conceptualised as reflecting a balance between the identity as a person and that of a patient resulting in a degree of equilibrium which was achieved through a degree of acceptance and positive expectations about the future. Subsequently this conceptualisation was operationalised using 40 items based upon prior qualitative work to reflect this definition. Psychometric testing then produced a solution with four factors reflecting patient identity, person identity, acceptance and expectations which were shown to have a robust factor structure and mostly good internal consistency as measured by the Cronbach’s alphas and corrected item-total correlations of the factors, with the acceptance subscale demonstrating slightly poorer internal reliability than the other three subscales. Association with existing measures was then assessed through correlations with measures of sense making (the brief IPQ) and quality of life (the IBDQ) indicating weak to moderate correlation strengths suggesting that whilst adaptation is associated with sense making and quality of life in IBD it is not synonymous and therefore measuring something unique. The present study also explored the relationship between the process of adaptation and illness outcomes. Specifically, the aim of the study was to investigate whether adaptation, as conceptualised in Matini and Ogden (2015) and then operationalised by the A-IBD, could predict the outcome of QoL in patients with IBD, as measured by the IBDQ/IBDQ-S. With regards to those patients without stomas, it was found that A-IBD scores significantly predicted QoL scores, with the four factors of the A-IBD accounting for 45.0% of the variance in IBDQ scores, as well as demonstrating a moderate effect size as evidenced by an adjusted R2 of .441. Assessing the factors of the A-IBD independently, it was found that both patient identity and person identity scores significantly contributed to IBDQ scores, with coefficients of 17.140 and -19.494, respectively. Acceptance and expectations meanwhile 115 were not found to significantly contribute to IBDQ scores, with coefficients of 3.214 and 4.066, respectively. With regards to those patients with stomas, it was found that A-IBD scores again significantly predicted QoL scores, with the four factors of the A-IBD accounting for 52.0% of the variance in IBDQ-S scores, as well as a medium effect size as evidenced by an adjusted R2 of .467. Assessing the factors of the A-IBD independently, in this case it was found that only patient identity was found to significantly contribute to IBDQ-S scores, with a coefficient of -25.729. Accordingly, person identity, acceptance and expectations were not found to significantly contribute to IBDQ-S scores, with coefficients of 5.296, -.067 and 6.631, respectively. When assessing to what degree, if at all, the A-IBD could predict IBDQ/S scores over and above BIPQ scores, it was found that the A-IBD scores helped explain a further 6.6% and 5.1% of the variance in IBDQ/S scores, respectively, than BIPQ scores alone. 3.5.2 Links to literature Where the previous study conceptualised the notion of adaptation (Matini & Ogden, 2015), the current study now operationalised the concept so as to make it measurable. In doing so, the findings from the current study which highlight the factors that constitute adaptation help to bridge the gap between the issue of the myriad of complex challenges that patients with IBD face, as outlined in Devlen et al.'s (2014) conceptual model of the impacts of IBD, with a solution as to how patients overcome these challenges. Up to this point, we largely understood the problems that IBD poses but little on how exactly patients evolve in light of these problems. 116 While the conceptualisation and operationalisation of adaptation to IBD that has been developed thus far differs, in some cases quite significantly, to other theorised conceptualisations of the construct in the IBD literature, it also finds a degree reflection in most of the thus far postulated conceptualisations. For example, Olbrisch and Ziegler's (1982) suggestion that disease-related adjustment in IBD was associated with patients' general level of self-esteem and social competence is particularly demarcated from its composition as proposed by the current study (and that of the previous chapter). However, Maunder and Esplen's (1999) and Voth and Sirois' (2009) propositions of 'adjustment' to IBD do share some similarities. In the former it was posited that adjustment was associated with illness uncertainty, loss and change, and suffering. Indeed, the conceptualisation of adaptation in the previous chapter, which then gave rise to the measure in this chapter, argued that patients seek to balance their patient identity with their person identity (two of the factors found to be associated with adaptation after the above factor analysis) in order to attempt to overcome the uncertainty that is inherent when living with the illness. Meanwhile, while in the latter poor adaptation was suggested to be largely attributed to self-blame, a factor that did not arise in the current study, it was also purported to be related to increased avoidant coping. Despite this thesis focusing on identity in the form of the patient identity and the person identity, these facets of illness identity are undoubtedly associated with certain instances of coping. For example, it is arguable that a patient scoring low on the person identity scale and thus not exerting themselves, not socialising, being hyper-vigilant with regards to their fatigue, and so forth, will demonstrate instances of behaviour that are typically associated with as emotionfocused or avoidant coping, and it is also probably likely that these behaviours will be predictive of poor adaptation as Voth and Sirois (2009) found. The results of the current study also support some other well-established findings in the chronic illness literature, primarily the finding that acceptance is a useful predictor of 117 QoL. While the current study found that it did not significantly contribute independently to QoL, as measured by the IBDQ, neither in those with stomas nor those without, when combined with the other factors as part of the overarching regression model, it aided in significantly predicting QoL. Its justification as an important factor despite its non-significant individual contribution is supported by research into the importance of acceptance on QoL in research on patients from multiple illness groups including those suffering from haemophiliarelated joint pain (Elander et al., 2013), cancer (Heydarnejad et al., 2011), diabetes (Lewko et al., 2007) and multiple sclerosis (Van Damme et al., 2016). Meanwhile, despite expectations again being non-significant in predicting both IBDQ and IBDQ-S scores, its inclusion as part of an overall significant model of adaptation that predicts QoL reflects the notion of 'positive illusions' from Taylor's Cognitive Adaptation Theory (Taylor, 1983) in being a critical part of adaptation. Finally, although the concept of identity and its relationship with adaptation has garnered the least attention in the literature of the emergent factors of the A-IBD, it has indeed been alluded to as a crucial part of the patient experience in chronic illness. For example, Moss-Morris' (2013) working model of adjustment to chronic illness mentions the potential for IBD to interfere with patients' identity or life roles which contributes to a disrupted emotional equilibrium and subsequently impaired QoL. 3.5.3 In summary Overall, the current study attempts to build on the previous study's conceptualisation of adaptation to IBD by operationalising the construct. As a result, thus far we have advanced the IBD literature, and arguably the wider chronic illness literature, to the point where we not only have an understanding of what constitutes adaptation to IBD which was previously very poorly conceptualised, but also can now begin to quantify these patients' level of adaptation to their illness. 118 Further to this, the current study builds on the story of this thesis by justifying the importance of understanding the process of adaptation to chronic illness, and specifically to IBD in this case. This justification is supported by this study's finding that it has some utility in predicting illness outcomes in IBD, with the current study focusing on the outcome of QoL as measured by the IBDQ. However, the narrative thus far, as established by Matini and Ogden (2015) is that adaptation to IBD has been conceptualised and operationalised as a balance between the person and patient identity along with acceptance and expectations, whereas the results from the present study alter this conceptualisation slightly. Whereas thus far it has been advocated that there should be equilibrium between the patient and person identities in order to achieve good adaptation, the findings outlined above indicate that in fact a positive, linear relationship between person identity and QoL and a negative, linear relationship between patient identity and QoL exists. As such, while our original conceptualisation of adaptation to IBD constituted a search for the 'new normal' by counterbalancing one's patient identity with one's person identity, rather it appears that we should simply encourage the person identity and inhibit the patient identity, and it is perhaps this encouragement of the person identity that helps restore equilibrium to the lives of those impact by IBD. Now that we have better ascertained what constitutes adaptation in IBD, how to measure it, and that it is a worthwhile paradigm by which to go about improving QoL in this illness group, the aim of this thesis will now be to answer the question of how patients reach a point of being adapted or maladapted. By investigating this, we should gain an understanding of what patients feel enables them to score high for those well adapted, and what prevents them from scoring higher for those less well adapted. Ultimately, it is hoped that these enabling or preventing factors are amenable to change, with a view to subsequent interventions that can target these factors and resultantly improve QoL. 119 3.5.4 Methodological limitations In terms of limitations of the study, it is arguable that our results are a product of both the recruitment method used (online support groups) and the large bias of UK participants. With regards to the former, the external validity of the A-IBD may be put under scrutiny since the sample of participants may not be representative of the wider IBD population. Meanwhile, with regards to the latter, of the 307 who were included in the final data analysis, 154 classified themselves as White British which again brings into the question the representativeness of the A-IBD with the wider IBD population. In addition, female participant representation (83.1%) far outweighed male participant representation (16.9%). The fact that the previous study was also represented mainly by female participants (14 females compared to nine males, i.e. 63.6% compared to 36.4%, respectively) somewhat allays concerns regarding the internal reliability of the measure since it was both formed (Chapter 2) and responded to (Chapter 3) by a predominantly female sample of patients. However, this gender bias does potentially raise concerns over the external validity of our findings regarding adaptation to IBD since it was conceptualised and operationalised on a largely female sample and thus may not be representative of the male IBD population. In this case, the concerns are again somewhat diminished by the findings of a female bias in autoimmune disease susceptibility in general (Ngo et al., 2014) as well as IBD prevalence statistics (Betteridge et al., 2013, Dahlhamer et al., 2016). It should be noted though that this female bias in IBD prevalence is typically a characteristic of Western IBD patients, again raising issues regarding the findings' representativeness as was highlighted above by the acknowledgement of a generally UK based sample. Another issue worth raising is that participants' diagnoses were self-reported and thus our results and conclusions are reliant on participants accurately and truthfully reporting this information. 120 Perhaps the most obviously questionable aspect to the methodology of the current study though was that the non-stoma group, which completed the IBDQ, far outnumbered the stoma group, which completed the IBDQ-S (by 266 participants to 41). However, this bias towards the non-stoma group was somewhat alleviated by both the data screening process and the subsequent regression results themselves. With regards to the former, multiple steps were taken to ensure the group met all the statistical assumptions necessary to carry out a multiple regression such as ensuring a sufficient sample size and assessing homoscedasticity and multicollinearity. Furthermore, while the results of the stoma group did differ from the non-stoma group in that person identity was not a significant contributor to IBDQ scores in the stoma group whereas it was in the non-stoma group, the remainder of the results were very similar, with patient identity being a significant contributor to IBDQ/IBDQ-S scores but not acceptance or expectations. As such, the combination of the data screening procedures and the similarity of the results should ameliorate any concerns regarding the disparity in size between the two sub groups. Another potential flaw of the study is that while the study establishes that A-IBD scores were significantly predictive of IBDQ/IBDQ-S scores, with both regression models demonstrating medium effect sizes, these effect sizes are merely representative of correlations and thus do not necessarily indicate causation. Furthermore, the adaptation formula that was decided upon based on the A-IBD and IBDQ/S regression models may be limited somewhat. First of all, only patient identity was a statistically significant contributor to the prediction of both IBDQ and IBDQ-S scores. Also, when predicting IBDQ-S scores, acceptance had virtually no impact. However, in both instances when all four subscales were included the regression model was highly significant and thus all subscales were retained. It should be noted though that perhaps the mechanisms 121 of adaptation are marginally different in those patients with IBD who have a stoma and this warrants further investigation. Finally, it could be argued that a 5-6% improvement in the prediction of IBDQ/S scores does not represent a significant improvement over merely using the BIPQ. However, it should be noted that, firstly, the BIPQ contained four more variables than the A-IBD and an increased number of independent variables tends to increase the percentage of variance of the dependent variable that is predicted. Secondly, while statistically perhaps this improvement is small, it is ultimately the impact on patient wellbeing that is important and according to this analysis, the A-IBD may help us improve our understanding on how to predict QoL and thus improve it. Finally, the current study did not carry out all stages typically involved with questionnaire validation, as outlined at the beginning of the chapter. For example, it is typically good practice to involve practice in questionnaire development to involve the intended end users, in this case the patients with IBD, and seek their views on item selection, item reduction, and opinions on face validity of the final measure. In addition, there was no involvement of IBD clinical experts in either item selection or item reduction which would provide feedback as to the content validity of the measure. The involvement of both could have given some insight into whether such a measure could be potentially beneficial based on any prior experience undergoing psychological intervention for the former, and administrating psychological interventions for the latter. However, repeated engagement with a patient pool of the magnitude of the one involved in this study as well as collaborating with IBD clinical experts far exceeded the scope of this thesis. While foregoing these methods of measure validation do raise some doubts as to the efficacy of the measure, carrying out these steps would have likely been too time consuming to be pragmatic in the context of this thesis. 122 3.5.5 Conclusion To conclude, this study presents a new 18 item scale to measure adaptation in those with IBD - the A-IBD - which shows mostly good reliability. This study also study provides support for the A-IBD as measuring something distinct from measures of sense making and QoL, as well as providing support for the A-IBD as an effective means of predicting QoL in patients with IBD based on their degree of adaptation to their illness. A qualitative study will now be carried out in Chapter 4 in order to understand the factors that patients feel encourage or inhibit their adaptation to IBD so as to guide therapeutic interventions in IBD towards the goal of being well adapted. 123 Chapter Four Study 3: Patients' explanations for their degree of adaptation: a qualitative study with a quantitative component 4.1 Overview The previous chapter sought to assess, firstly, the degree to which the new measure of adaptation to IBD (the A-IBD) was associated with existing measures of sense making (the Brief IPQ) and health outcomes (the IBDQ). The chapter also assessed the ability of the AIBD to predict QoL over and above the predictive ability of the BIPQ. The results indicated that the A-IBD did indeed measure something unique compared to the BIPQ and IBDQ and that the A-IBD predicted a further 6.6% and 5.1% of the variance in IBDQ and IBDQ-S scores, respectively, than the BIPQ alone. These results suggested that the A-IBD has utility in measuring adaptation and that a state of high adaptation is indeed beneficial in terms of QoL and therefore to be aimed for if possible. This next chapter will therefore explore the underlying factors that contribute to high and low adaptation as measured by the A-IBD. This will be achieved by interviewing a sample of those patients with IBD who were positioned in the top and bottom 25% of scorers on the A-IBD from Study 3 (Chapter 4), to explore the reasoning behind their prior responses with regards to the factors they felt promoted or inhibited their adaptation. This chapter will also explore the notion of whether adaptation should be regarded as either a state or trait based on the degree of variation in participants' A-IBD scores between Study 2 (Chapter 3, T1) and the current study (T2). 124 4.2 Introduction The process of adaptation to chronic illness has garnered little attention in the chronic illness literature to date. In its place, much work has been done on underlying psychological processes such as research into theories of coping (Lazarus & Folkman, 1984; Moos & Schaefer, 1984) and sense making in the form of Taylor's Cognitive Adaptation Theory (Taylor, 1983) and Leventhal et al.'s (1984) Self-Regulatory Model. While these frameworks have proven their utility in predicting health outcomes across a number of illness groups such as those suffering from hypertension (Meyer et al., 1985), Chronic Fatigue Syndrome (MossMorris et al., 1996) and cancer (Czajkowska et al., 2013; Tomich & Helgeson, 2006), they fail to account for the lived, biopsychosocial experience that is characteristic of chronic illness. Due to the complex physical, social and emotional factors associated with living with IBD (Gearry et al., 2006; Czuber-Dochan et al., 2013; Vidal et al., 2008) along with the fact that the disease is characterised by an unclear aetiology (Fiocchi, 1998) and a limited, potentially dangerous treatment protocol (Stallmach et al., 2010), understanding adaptation to IBD is particularly important and yet thus far lacking. While more recent research such as that of Moss-Morris's (2013) working model of adjustment to chronic illness has built on the aforementioned frameworks in that it attempts to bring together the aforementioned psychological processes together with emotional and behavioural factors, the concept of adaptation has remained poorly conceptualised in terms of how this framework applies to specific illness groups. Through Studies 1 and 2 of the current thesis, this gap in the literature has now been addressed with both the conceptualisation of IBD and its subsequent operationalisation in a new measure of adaptation focusing on the experience of the patients with a particular focus 125 on the notions of patient identity, person identity, expectations and acceptance. At this point, however, the question remained as to whether this understanding and operationalisation of the process of adaptation could provide utility in its ability to predict health outcomes in the form of a subjective, IBD-specific measure of IBD - the Inflammatory Bowel Disease Questionnaire (IBDQ; Guyatt et al., 1989). Through the analysis carried out in Study 3 (Chapter 4), we are able to confirm that not only does the A-IBD sufficiently predict subsequent patient HRQoL, but is also a construct that is distinct from both illness representations, as measured by the Brief Illness Perceptions Questionnaire (BIPQ), as well as QoL itself as measured by the IBDQ, and is thus not synonymous with either of these constructs. With an understanding of what constitutes adaptation to IBD, the focus now moves to gaining an understanding of the contextual antecedents of those that present as adapted or maladapted. With the development of this knowledge, it is hoped that future interventions aimed at improving patients' degree of adaptation to their illness will be targeted at the appropriate factors that engender this adaptation. To date, psychological interventions aimed at treating IBD through the application of a range of strategies including psychotherapy, counselling, and cognitive behavioural therapy have all proven to lack in long-term efficacy with regards to their effect on QoL, (Timmer et al., 2011; Smith et al., 2002; Mikocka-Walus et al., 2016). However, upon successful identification of the antecedents of adaptation, ideally we should observe an improvement in the efficacy of future interventions that target these specific factors. In line with this ultimate goal, the primary aim of the current study therefore was to identify those factors that patients themselves feel promote or inhibit their level of adaptation to their illness. Specifically, this study will focus on those patients who previously presented as being either particularly high or low in adaptation from Study 2 (Chapter 3). 126 How adaptation scores are determined as high or low for the current study will be explained in the following section. Adaptation is largely agreed upon in the chronic illness literature as a dynamic concept, continuously evolving and unpredictable and dependent upon a myriad of intrapersonal (biopsychological), interpersonal (sociocultural) and extrapersonal (environmental) factors (Linveh, 2001). So far however, this thesis has only used a cross sectional design and has therefore only measured adaptation at a single time point. The second aim of this study was therefore to explore the extent to which adaptation to IBD, as measured by the A-IBD, changes over time and can therefore be considered either a stable trait or a more dynamic state. In line with this, a sub sample of participants from Study 2 were followed up in the current study between 10 and 16 months after their baseline measure and their adaptation measured for a second time to assess any degree of change. 4.3 Method 4.3.1 Design A qualitative and quantitative design was used with in-depth, semi-structured interviews and a repeat assessment of the A-IBD. 4.3.2 Sample Participants for this study were recruited by identifying those scoring in the top and bottom 25% of scorers on the A-IBD from Study 2 (Chapter 3). In line with the criteria for participant recruitment in this prior study, participants were required to be at least 16 years of age and capable of providing informed consent. However, only those participants living in the UK were invited to take part in the current interview study. 16 participants were recruited upon which point saturation was reached: High adaptation (n = 8; six females, mean age = 127 33.0 years, range = 23-47 years; two males, mean age = 33.0 years, range = 24-42 years; mean time since diagnosis = 100 months/8 years), low adaptation (n = 8; six females, mean age = 41.0 years, range = 24-57 years; two males, mean age = 30.0 years, range = 24-36 years; mean time since diagnosis = 145 months/12 years). All participants were given a pseudonym to ensure anonymity. Participant demographics are presented in Table 8 below. 4.3.3 Classifying degree of adaptation Participants were selected for the current study based on their responses to the A-IBD in Study 2 (Chapter 3). Those scoring in the bottom 25% of scorers (up to and including an A-IBD score of 4.92) were classified as low adapters and those scoring in the top 25% of scorers (including and above an A-IBD score of 7.30) were classified as high adapters. 4.3.4 Change in adaptation Participants' adaptation scores, according to the A-IBD, from the current study (T2) were compared to their initial A-IBD scores from Study 2 (Chapter 3, T1) to assess whether adaptation to IBD was a state or trait. Accordingly, Table 8 below states each participants' T1 and T2 A-IBD scores, classified as showing 'no change' if T2 scores were within ±1 of T1 scores, as 'improved' if T2 scores were ≥1 greater than T1 scores and as 'deteriorated' if T2 scores were ≥1 lower than T1 scores. Based on this criteria, it can be seen that six participants showed no change in adaptation, three participants improved and six participants deteriorated. 128 Table 8: Participant demographics Pseudonym Sex Age (years) Diagnosis Time since diagnosis (months) A-IBD score T1 A-IBD score T2 Change in adaptation (T1/T2) Brian M 24 UC 74 9.57 7.61 Deteriorated Rhian F 24 CD 59 4.65 6.95 Improved Elaine F 23 CD 52 9.17 4.20 Deteriorated Zara F 24 UC 44 10.83 7.50 Deteriorated Bob M 24 UC 69 4.23 5.13 No change Janet F 36 CD 100 9.28 7.20 Deteriorated Jason M 42 CD 297 9.28 9.86 No change Daniel M 36 CD 130 4.10 9.03 Improved Stephanie F 40 CD 292 3.73 4.94 Improved Joanna F 44 UC 46 8.35 6.53 Deteriorated Jackie F 47 CD 112 8.88 9.15 No change Katie F 45 CD 376 4.00 3.40 No change Sharon F 57 CD 53 3.83 3.45 No change Philippa F 24 CD 71 9.13 6.84 Deteriorated Sarah F 52 CD 107 4.48 4.10 No change Diane F 28 UC 70 4.85 5.20 No change 129 4.3.5 Procedure Participants were followed up between 10 and 16 months from the time of the first interview (Study 2, Chapter 3). Participants from the top and bottom 25% of scorers on the A-IBD at baseline and living in the UK were e-mailed to inform them of their status as a participant of interest for the current study. All interviews were conducted in English, following a semi-structured interview schedule (See Appendix M) over the telephone at a mutually convenient time and were all carried out, audio taped, transcribed verbatim by the principal investigator and then stored securely on the University servers according to the Data Protection Act 1998. Telephone interviews were preferred to face to face interviews due primarily to pragmatism since it would allow for participants from all over the UK to participate, as well as allowing participants to share their opinions in a relatively anonymous fashion. Interviews lasted on average between 30 to 45 minutes and took place over the course of three weeks in August 2016. Once the interviews had been conducted, participants were granted the opportunity to ask questions they had regarding their participation or to reflect on their experience. All participants were given a verbal debriefing to explain the nature of the study they had been involved in. Ethical approval was obtained from the University Ethics Committee (See Appendix N) 4.3.6 Interview Schedule The same interview schedule was used for both the CD and UC participants. The first section of the interview covered briefly the participants' history with IBD (When they were diagnosed; was the diagnosis a lengthy process), followed by a discussion of the major difficulties the participants had faced emotionally, psychologically and behaviourally with regards to their illness. Once a rapport had been established, participants were then interviewed regarding the answers they had given to the A-IBD from Study 2 (Chapter 3) 130 based on the four subscales of the measure (patient identity, person identity, acceptance and expectations). Questions revolved around what factors the participants felt contributed to their scores on each of the subscales. The interview schedule was piloted with two patients with IBD prior to the study. 4.3.7 Data Analysis As was the case with Study 1 (Chapter 2), the interviews were analysed using thematic analysis. Within this analytical framework, an inductive, 'bottom-up', approach was taken as our analysis was not based on any previous research into aspects that contribute to adaptation, rather we sought to generate phenomenological data from which our themes could be developed. Furthermore, an essentialist theoretical perspective was chosen as our focus was on the experiences, meanings and reality of the participants. The interview transcriptions were each read through twice in order to become familiar with the data. Salient information that related to the lived experience of IBD was then coded and collated and sorted into a number of prospective themes that seemed to represent the data. Themes and subthemes were adjusted to include only those that could be supported by illustrative quotes from the interviews. 4.4 Results Upon reviewing and discussing the participants' results from the A-IBD, differences could be seen between the high and low adapters with regards to three core themes, namely their level of 'engagement', 'resilience' and certain 'contingencies'. These themes will now be elaborated on with the use of illustrative quotes and are outlined below in Table 9. 131 Table 9: Themes and subthemes of thematic analysis Themes 1. Engagement 2. Resilience 3. Contingencies Sub-themes i. Fulfilment ii. Honesty iii. Openness i. Minimising ii. Reframing negativity iii. Realising unconstructive cognitions i. Disease factors ii. Social factors Theme 1: Engagement The first significant way by which participants who scored highly on the A-IBD differed from those who scored at the lower end of the scale was in terms of their propensity to engage more in three specific areas of their day to day life. These participants demonstrated more willingness to engage in activities and hobbies they enjoyed or even loved so as to gain a sense of 'fulfilment', 'honesty' in terms of their own assessment of their levels of perceived fatigue and whether they could push through this barrier of fatigue, and 'openness' when it came to communicating the current situation of their illness with others and sharing their disease experiences with the wider community. 132 Subtheme 1: Fulfilment Almost every participant in the high adaptation group spoke of their desire to simply do things they enjoyed in spite of their illness. For some these activities also served the purpose of distracting themselves from their illness and preventing possible rumination and complacency as one participant described who had rediscovered the importance of playing music in her life: "It was actually after the diagnosis I thought you know what I need to start doing this again, and that probably helped, it kind of made me go back and start playing and that gave me that focus and I could get away from sitting there" (Janet, 36, CD) For others the notion of fulfilment served as a protective factor against compromising one's identity: "I think it’s really important to let people realise that the thing that they enjoyed when they weren’t feeling ill, so whether that’s looking at some nice countryside or playing with the dog or watching their favourite actor in a film on TV or whatever, they can still enjoy even if their guts are giving them an absolutely horrible time, it hasn’t fundamentally changed you as a person" (Joanna, 44, UC) Finally, one participant whose adaptation score rose significantly between the initial and follow up A-IBD assessment demonstrated perhaps the key difference between higher and lower scorers in terms of this domain of adaptation: the importance on seeking fulfilment in the face of fear from potential disease repercussions, be they symptoms or injury to areas on the patient's body that had been operated on due to their illness: "I could just sit there and not try, I could try and fail, but just sort of thinking well try it and if you fail fair enough but try it, so like I started kickboxing, I started that last week, 133 and so lots of people were like oh kickboxing but what about your stoma and that and I think well yes that plays a part in my thought process but let’s see how I go first" (Daniel, 36, CD) In contrast, Elaine, whose adaptation score significantly fell from high to low was seemingly increasingly concerned with returning to her hobbies of football and martial arts for fear of worsening her symptoms. Crucially, she admitted that what was preventing her from engaging in these activities was likely mental in nature than physical: "I used to do a lot football, I used to do a lot of martial arts before I got diagnosed...I’m just too scared to do them...it would probably be alright after a certain amount of time but mentally I just can’t do it... I don’t wanna make it worse, I just don’t wanna make myself even iller than I already am" (Elaine, 23, CD) Other examples of this type of lack of engagement from low scorers was demonstrated by one participant who, despite on one hand having previously described her inability to get out the house had also talked about how she was indeed successful in venturing out the house and being active with the assistance of a psychologist at a previous point in time, but had now reverted to limiting herself to sedentary hobbies at home again: "My hobbies are sedentary which is knitting, reading and playing games. I used to love cycling but I mean I haven’t been able to do that for years, so in that case I don’t " (Sharon, 57, CD) Crucially, this was by the participant's own admission linked, as with the previous participant, to psychological reasons than to any current bout of disease activity: "No my illness hasn’t got worse, I think my feelings about it have" (Sharon, 57, CD) 134 Subtheme 2: Honesty Fatigue was perhaps the most common complaint among all participants interviewed. However, participants scoring high in adaptation demonstrated a distinct ability to honestly analyse their fatigue and determine when they were pushing their boundaries to the point that their exertion would become detrimental to their illness: "That’s something that only really you can work out, that’s something that a doctor won’t be able to tell you how to do and research probably won’t be able to accurately indicate for every individual how to do it, it’s something you need to learn for yourself...it’s just about learning the very subtle difference between normal fatigue and like a flare up induced fatigue...if I gave into fatigue every single time then I would end up housebound again, but on the other side of the coin, if I pushed through all of the fatigue I feel then I would make myself ill more often, so there is a balance there " (Brian, 24, UC) Similarly, one participant demonstrated this honesty with regards to attributing how much of his fatigue was likely due to reasons other than his disease: "I am the run ragged father of a 10 year old and a 7 year old and I've got a pretty big job here...I’m a bit fatigued, the attributableness of that fatigue I have got no idea, 3% of it may be down to the fact that I have a chronic inflammatory disease, 97% of it is I’ve got two kids, so yes I’m tired but I really don’t think very much of it is to do with the fact I’m unwell" (Jason, 42, CD) Once again, Daniel's metamorphosis from low adaptation to high adaptation was reflected in his ability to challenge his fatigue and come to the realisation that pushing one's perceived limits in terms of fatigue can provide positive reinforcement: 135 "This week’s session was a lot more full on and yeah I didn’t come out thinking oh my God I’m exhausted if anything I came out feeling slightly more energised thinking I wanna do more I wanna do something else now...I think it may be partly because you stop yourself doing anything because you’re so ill so you forget what that feeling is like when you do some form of exercise" (Daniel, 36, CD) On the other hand, participants scoring low in adaptation tended to demonstrate a sense of helplessness and resignation to their fatigue as opposed to an honesty as to their ability to challenge it, as evidenced succinctly by one participant in particular: "I just think that it’s just one of them things, I mean with Crohn’s one of the symptoms is fatigue and I’ve just got to put up with it really" (Katie, 45, CD) This same participant then went on to characterise the negative reaction to fatigue following exertion that was common of low adaptation scorers: "I can get up sometimes really early and the next day it’ll kill me, it’s like I’ve been walking recently and I mean I’ve been aching now for a couple of days" (Katie, 45, CD) Subtheme 3: Openness The final way that participants scoring highly on the A-IBD differed to those who score lower in terms of their degree of engagement was with regards to their tendency to be more open about their illness. The ways in which this openness was displayed varied, with one participant explaining how it was important to her to be transparent with friends when her illness intervenes with plans: "I am (open) but only if it needs to be stated I think, if something’s happening and I have to back track or back out or change plans I wouldn’t just do it and leave people wondering, I would explain nicely" (Jackie, 47, CD) 136 This openness was also shown to be valuable in the workplace, with one participant describing that, despite her initial anxiety at being negatively perceived for announcing her illness to her boss, a positive outcome occurred as a result of her disclosure during a particularly bad period of disease activity: "He was very good and sort of said well you know look after yourself, if you need to work from home that’s fine let me know how you’re doing, and he’s not really said anything about it since but I don’t worry that he’s sort of thinking oh my god have I employed this person who’s just gonna be a malingerer or have a lot of time off sick or whatever" (Joanna, 44, UC) Most striking though was the willingness of some of the participants scoring high in adaptation to be open about their illness with the general public and the positive outcomes this engagement had on their perspective on life with IBD. For one participant this opportunity arose from writing a book about his experiences with his illness: "It was when I sort of published my book and people realised the extent of the illness a lot of people got in touch and sort of started sharing their own stories, and it was people I’d never expect sort of people from school who were saying they had confidence issues and they had to take antidepressants and things...you start to realise that really we all have weaknesses within ourselves that we all have to deal with and that just makes it easier to accept" (Brian, 24, UC) Meanwhile, one participant scoring at the lower end of the A-IBD spoke of how she struggled to reconcile the fact that she understood keeping her friends and others "at arm's length" was counter-productive with the fact that at the same time these other people, in her opinion, could never fully understand what she was going through with the illness due to its 'invisible' nature: 137 "Obviously I know I keep people at bay and at arm’s length so I know I need to change that or I’d like to, but then from their point of view it is often referred to as an invisible disease so obviously when I go out they’re only seeing me when I’m feeling OK and with loads of makeup and everything so they don’t really believe that there’s anything wrong" (Stephanie, 40, CD) Differences in the levels to which those demonstrating high and low adaptation engaged in all aspects of their life, despite the burdens placed on them by their illness, was therefore described in a number of ways. Throughout the accounts, these differences revolved around the degree to which the highly adapted participants more often sought fulfilment in their lives by continuing to engage with their passions, by being honest with their fatigue and continuing to engage when they deemed it sensible and not detrimental to their ongoing wellbeing, and by being open with friends and family as to their limitations and sharing their experiences with others. Theme 2: Resilience Another stark difference was also observed in the way in which participants who scored highly on the A-IBD demonstrated a seemingly greater ability to psychologically and emotionally respond to the often daily challenges that IBD posed. This was exhibited by the high scorers in three main ways, namely through 'minimising', 'reframing negativity' and 'realising unconstructive cognitions'. Subtheme 1: Minimising One way in which the high scorers demonstrated this level of psychological and emotional control was by minimising. For example, for one participant, evidence of this 138 ability came to the fore when they sat out a boat trip for the day while on a family holiday due a lack of toilet access which carried with it the potential for negative affect: "That wasn’t heart breaking, that wasn’t “Oh it’s not fair”, I didn’t think or feel any of those things, it’s just they couldn’t find a boat that had a loo so I said in that case I’m gonna have to be out I’m afraid, but that’s fine you go off and you have a great time" (Jackie, 47, CD) For another participant this ability to minimise was evident when they described how they had felt temporarily disheartened by a flare up of their illness and the associated fatigue. Crucially though they were able to rationalise the symptoms as part of the ambiguous nature of the disease despite their best attempts at preventing disease recurrence: "I've had really good compliance with my meds, I have been working quite hard but I haven't been really running myself into the ground, I didn't expect to get a bit ill again and so that has brought it back into focus for me, and it's just kind of from time to you are going to feel fatigue and you carry on as much as you can but you can't deny that it's going to happen" (Joanna, 44, UC) One participant extended this notion of minimising to both his positive achievements in life as well as his negative experiences with the disease, a philosophical mindset that appeared to help him better deal with symptoms such as pain: "There's this famous phrase about 'this too shall pass'. I've got it on every notebook I have at work...it's a two way thing right, all of those triumphs well they're going to go away at some point and be meaningless at the end of it, but it's also true for pain, eventually you will come to an accommodation with it so this too shall pass" (Jason, 42, CD) 139 In contrast to the above responses, one participant from the low scorers clearly found it far more difficult to come to terms with the fact that sometimes the disease would flare up without a logical cause for doing so despite being on medication, thus feeling there was no hope for the future: "It feels hopeless because at the moment there is no cure and there is no medication that helps stop...like I'm on Asacol which is to help stop the flares but it doesn't actually completely stop them, if I eat the wrong thing or just my body decides to have one I'll just have a flare, sometimes I can't even find a reason why I'm having one" (Sharon, 57, CD) This same participant then also demonstrated the difficulties that her and other low scorers often showed with regards to facing failures during their disease experience and how that cycle of negative reinforcement was difficult to break out of: "I need to lose weight and then every time I lose it I seem to get ill and then put it all back on again so it never happens and it just does feel like this is it, this is what I've got for the rest of my life however long or short it may be" (Sharon, 57, CD) Subtheme 2: Reframing A sizeable number of the participants also alluded to an ability to reframe potential negativity that may come about from living with IBD. Occasionally these allusions were associated with dealing with the physical aspects of life with the illness but more often participants spoke on a more metaphysical and philosophical level about living life with IBD and how they could rationalise and find positives from their experience. To highlight an example of the former, when one participant was confronted on the prospect of further IBDrelated surgery in their future she described how she would welcome the often-feared idea of living with a stoma since it would ultimately improve her health: 140 "These days I can be quite flippant and say if they took me in tomorrow and gave me a colostomy bag I'd be grateful because it would make my life a whole lot better. If it happens, it happens." (Janet, 36, CD) Others spoke of how they were able to reframe the negativity associated with periods of ill health as a "test" and thus something that they could challenge and potentially overcome: "Even if it’s harder than what you expected it to be then that’s just another part of the test, that’s just something you’ve got to accept and apply yourself to work around" (Brian, 24, UC) At other times, participants reframed the inherent negativity of their illness through rationalisation in the form of being mindful of the likely worse situations that others find themselves in: "I consider myself to have had an easy ride with Crohn’s even though actually I probably haven’t retrospectively, and in the context of 90% of the world’s population I am in as good as if not better place than anyone else...I am a relatively lucky human being" (Jason, 42, CD) Meanwhile, Daniel's change in adaptation score was reflected when he spoke of the potential for disappointment while dealing with a post-operation wound that wouldn't fully heal, but how he had found a way in the interim period to instil a feeling of purpose in himself by raising money for charity: "I bought myself a chin up bar and I’m basically approaching people to sponsor me so that I train throughout and then see in say New Year’s Eve in December people have sponsored me for however many pull ups…so that’s helping me keep some sort of focus on 141 feeling like I’m doing something... I’m at least doing something that’s, one, helping my fitness and, two, driving me to sort of say you know what again I’m not gonna let this beat me " (Daniel, 36, CD) Others still reflected on the positive changes they had seen in themselves due to living with their illness. For one female participant this was explained in terms of her ability to manage stress: "It’s made me a lot more I suppose chilled out about not trying to control things I can’t control, so for instance I used to get really, really annoyed in traffic jams and actually I’ve found since I sort of got ill and I am a slightly different person now, I’m much less uptight about it" (Joanna, 44, UC) Subtheme 3: Realising unconstructive cognitions Finally, participants who scored highly on the A-IBD demonstrated resilience by showing adeptness at recognising and altering thoughts and emotions that they acknowledged would be detrimental to both their physical and mental health as it pertained to their illness. In light of this, Zara described how important it was to "snap back" when she recognised this pattern of thinking in response to symptoms: "Everyone goes through an angry stage at some point in their illness, like I said sometimes I get frustrated...but it doesn’t take me long to kind of snap back with my mindset" (Zara, 24, UC) Similarly, ruminating on the possibility of future symptom occurrence was also a challenge. One participant in particular talked about how it was more the threat of potential embarrassment from symptoms occurring in public, the anxiety that had caused him and its 142 subsequent repercussions on his willingness to leave the house that was important for him to confront and recognise as irrational: "I remember quite distinctly I went to town for the first time after recovering from my symptoms and I started to get anxious and there was no real reason behind it and I got angry with myself because I knew I was letting myself slip into bad habits again, so I just basically told myself to get a grip and realise that my anxiety was irrational and I’ve done it before and I can do it again, and that anger actually helped me distract myself from the anxiety... it was almost an irrational level of anxiety, because the anxiety stems from not wanting to open your bowels in public but like the worst case scenario has happened and it’s actually not as bad as you’ve built it up in your head" (Brian, 24, UC) It is important to note that this same participant also acknowledged how the anger he spoke of could potentially be deleterious and that he rather used this emotion to channel into his hobby of power lifting: "It depends to what extent you’re angry and frustrated, so if you’re letting it physically cause you stress then yeah that can reinforce symptoms, it can reinforce bad habits and things, but you can use anger and frustration positively, so almost turn it into eustress rather than distress" (Brian, 24,UC) Retrospectively, another participant spoke of how he was at one time literally obsessed with assessing his disease activity and explained how this wasn't worthwhile: "Every day was Crohn’s and then I’d come home and do my own bloody HBI (Harvey Bradshaw Index) and then start pouring over my spreadsheet and wellness and whether I’m on a downward trend or an upward trend...the one thing that I didn’t analyse on top of that was how obsessed was I with my disease that day and then did it take up every 143 waking minute worrying about whether I would need that hemicolectomy or whatever" (Jason, 42, CD) This notion of obsession links to the final way in which participants who scored high in adaptation demonstrated an ability to recognise unconstructive cognitions and that was in relation to being able to resist being mentally 'consumed' by the illness as Daniel described it, further evidencing his transition from low to high adaptation: "It’s quite easy to sort of sit there and just sort of let it consume you and think that that’s it, because I did that and I did it lots of times" (Daniel, 36, CD) For another participant it was the explicit desire to not be 'defined' by the illness, having felt like she had been so in the past: "I don’t want to let it define me again, if it slows me down every now and again then I think I can sort of accept that but I don’t want it to be everything that I am again because that wasn’t very nice...I’ll get annoyed about it but I’m absolutely willing and sort of wanting that to be just a temporary thing rather than thinking oh this is how it’s gonna be forever now and I’m trying to think this is a little cloud coming across sunshine" (Joanna, 44, UC) Differences in the degree to which those demonstrating high and low adaptation demonstrated an ability to remain resilient despite their illness was therefore alluded to in three distinct ways. Highly adapted participants frequently demonstrated this ability by minimising the negative affect associated with the limitations and ambiguousness in terms of symptom onset associated with their illness, by reframing the inherent negativity of living with IBD and finding positives in the situation, and by better recognising negative thought patterns when they were beginning to manifest. 144 Theme 3: Contingencies The final theme that emerged from the interviews was that of 'contingencies'. While it was evident that the higher and lower scorers on the A-IBD could be differentiated by their willingness to engage and their psychological and emotional resilience, there were undeniably certain contingencies that need to be taken into account which may also differentiate the two groups. These contingencies can be categorised into 'disease factors' and 'social factors'. Subtheme 1: Disease factors A common issue for those scoring lower on the A-IBD was the existence of often physical, but also psychological comorbidities that further complicated living with IBD, arguably interfering with the process of adapting to their life as a patient with IBD. For example, one participant spoke of how she had developed chronic kidney disease as a result of one of her medications and the psychological impact this had on her: "the doctors should have picked up on it sooner rather than it be permanent, and at the time I was 22 and I’d had Crohn’s for 8 years at that stage, so being told again that I’ve another lifelong condition that I didn’t cause either, it’s just those kind of very negative things, you know, why me?" (Rhian, 24, CD) While fatigue was almost unanimously an issue for participants, be they higher or lower scorers on the A-IBD, for one participant it was the extra fatigue that was a consequence of his arthritis that was compounding the difficulties of life with IBD and the challenge of engaging despite fatigue in particular: 145 "My arthritis has sort of just continuously gotten worse, or at least has been a constant thing rather than before when it had flared up a couple of times. Since I started working that has just sort of drained a lot more of my energy than what I was prepared" (Bob, 24, UC) Others meanwhile had to contend with simultaneous physical and psychological comorbidities over and above their IBD, with one participant describing her difficulties with not only fibromyalgia but also depression: "Well depression is the main thing, I mean it’s a really big thing with it and that comes with the Fibromyalgia as well because I mean I’ve sat in my daughter’s office crying, I’m 57 and I feel like I’m already 90 and that’s because of all the things that I’ve got wrong" (Sharon, 57, CD) While other such as Daniel, while not suffering from depression as such, reflected on the difficulties of dealing with the psychological side effects of his previous medication that he took before making his stoma permanent which was likely a factor in his originally low score on the I-ABD: "I was on Humira for a while and what I used to find with that is if I took it, the next day I’d just feel a bit…I don’t want to use the term depressed because it’s not that bad but I just didn’t feel right and then sometimes what would happen is you’d be in that sort of mood and then something would happen that day and it might just be something really tiny but it affects you 10X more, whether it’s something that makes you angry, makes you upset…it didn’t seem to work for the happy side " (Daniel, 36, CD) Finally, sometimes it was the consequences of previous surgery that were clearly limiting the engagement of participants: 146 "Because of the amount of scar tissue I have from the surgeries that I’ve had means that I can’t move around as much as I used to be able to, I can’t stand up for any huge length of time or walk any great distance, as a result of that I’m not as active as I used to be and I’ve put on weight, that sort of thing so yeah to me it’s a bit of a bit of a vicious thing that I don’t know how to get out of" (Sarah, 52, CD) Subtheme 2: Social factors In conjunction with the added disease factors that lower scorers on the A-IBD tended to be faced with, they also communicated a range of social factors that are likely to have inhibited adaptation to their illness. In some instances it was a lack of family support and understanding that was cited, as was the case with Elaine who had reverted from being in the high scoring A-IBD group to the low scorers: "Even though I’ve had this disease for 4 years they don’t understand what the symptoms are, they don’t understand the fact that yeah you can get tired and you can get all these symptoms very quickly and sometimes I’ve had arguments about it as well, especially with my parents, about the fact that I can’t do a lot of stuff because of my Crohn’s but they just don’t seem to understand" (Elaine, 23, CD) Another participant described how, on top of dealing with depression, she had gone through a divorce with her previous husband of 18 years prior to bowel resection surgery which meant she would require a temporary ostomy bag: "His reaction was if I was gonna have a bag he was gonna leave me so I did it (ended the relationship) before he did... it did get me down quite a lot" (Katie, 45, CD) One participant expressed her difficulties with managing her symptoms while at work which had triggered anxiety issues and subsequently led to her losing her job: 147 "I was working like a busy store and the facilities there weren’t ideal because they didn’t really have for example a disabled toilet for the staff, there was only one for the customers which was obviously busy or unclean, so I sort of struggled with my working environment, that then triggered the anxiety because I worried of sharing with quite a young work force, so that then triggered the anxiety and then I lost my job... they didn’t really make reasonable adjustments so in the end they sacked me for incapability or incapacity" (Diane, 28, UC) Undoubtedly the aforementioned disease factors likely have repercussions on the thus far listed social problems many of the lower scorers on the A-IBD faced, but perhaps none were as directly linked as one participant whose 12-13 year long, ongoing fistula issue subsequently led to both her inability to work and simultaneously the breakdown of her relationship at the time, culminating in often being isolated from others: "I keep everyone at bay, I just don't like my body and I find for me managing it takes up a lot of my time...I feel with the fistula I constantly feel dirty and not so much from the scars, I really don't care about those, but it's more I can't trust my body to behave socially" (Stephanie, 40, CD) Through all of these issues however, it should be noted that participants were often aware of the detrimental effects that becoming complacent with their situations could potentially have, with the above participant in particular explaining how she was trying to make a conscious effort to begin establishing meaningful relationships and interacting socially once again by moving closer to her family and beginning a degree in psychology: "I’m going back to study to be around more like-minded people than what I have here...I’m hoping with my family I won’t be spending as much time on my own, and I’ve always tried to do courses, long distance learning, but I miss that interaction with people so 148 I’m hoping through this course I will meet more people and spend more time with people. Apparently if I practice being around people it will get easier" (Stephanie, 40, CD) The differences between the high and low adapters could therefore be attributed somewhat to certain disease related social contingencies. In this respect, participants demonstrating lower adaptation commonly raised issues regarding physical and psychological comorbidities, as well as certain side-effects of medication, which impact their ability to function, along with feelings of a lack of support from friends, family and partners. 4.5 Discussion 4.5.1 Summary of findings - thematic analysis The present study explored primarily the factors associated with high adaptation compared to low adaptation, with those demonstrating high and low adaptation being classified as the top and bottom 25% of scorers on the A-IBD from Study 2 (Chapter 3). Thematic analysis highlighted three core themes relating to 'engagement', 'resilience', and 'contingencies'. With regards to the core theme of engagement, three emergent subthemes were found including 'fulfilment', 'honesty' and 'openness'. With regards to the core theme of resilience, another three subthemes were found including 'minimising', 'reframing negativity' and 'realising unconstructive cognitions'. Finally, with regards to the core theme of contingencies, two emergent subthemes were found including 'disease factors' and 'social factors' 4.5.2 Links to literature - thematic analysis The emergent themes from the current study find reflection in multiple aspects of the chronic illness literature, as well as IBD-specific literature. For example, with regards to the subtheme of 'openness', research has indicated that the onset of chronic illness is consistently 149 associated with decreased openness (Glavic et al., 2014; Jokela et al., 2015), which in turn may impact QoL via enhanced support systems (Schirm, 2012). For instance, one highly adapted participant indeed explicitly mentioned the positive impact he had experienced from publishing a book on his experiences with IBD. Crucially, the current study's findings, in particular the theme of 'honesty' highlighted notions of self-care and patient knowledge. This was demonstrated by the highly adapted participants' better understanding and honesty as it pertained to their fatigue and when they should limit themselves accordingly to protect their health, a facet of self-care that was raised in a recent qualitative study focused on self-care in patients with IBD (Wickman et al., 2016). Similarly, highly adapted participants were honest in terms of their ability to exert themselves with regards to getting out the house or exercising, for example, the latter of which has been shown to provide psychological benefit in IBD (Bilski et al., 2013) which may relate in part to these participants' overall greater QoL than less adapted participants. In addition, the theme of 'resilience', demonstrated particularly by the higher adapted participants, has been associated with improved HRQoL in the IBD literature (Hadianfard et al., 2015; Lanagran et al., 2016), again supporting the notion that these patients should report higher subjective QoL. Resilience has also repeatedly been referred to as a method of adaptive coping that enables those facing chronic stressors to strive (Cicchetti & Rogosch, 2009; Hopwood & Treloar, 2008) as evidenced by the participants in the current study when faced with the challenges of IBD. Finally, the 'contingencies' theme echoes the sentiment found in much research on the correlates of diminished QoL stating the deleterious effects of increased disease activity and poor social support (Kalafteli et al., 2013; Katz et al., 2016). These concerns were mostly highlighted by those demonstrating a lower degree of adaptation which is in line with these patients' generally lower level of QoL. 150 4.5.3 Summary of findings - adaptation - state or trait? The current study also investigated within this sub sample of 16 patients with IBD whether the concept of adaptation to IBD is dynamic, i.e. whether it is a state or trait. In this instance it was found that only two participants between T1 (Study 2, Chapter 3) and T2 (the current study) switched between the top and bottom 25% of scorers on the A-IBD (based on the scores from Study 2). Specifically, six participants displayed no change in their degree of adaptation, three participants improved in adaptation and six deteriorated. As such, while some participants demonstrated a stable adaptation score between T1 and T2, adaptation was proven to be a dynamic concept due to the variability in a number of the participants' A-IBD scores. 4.5.4 Links to literature - adaptation - state or trait? The comparison of the T1 and T2 A-IBD results from the current study also echo much of the literature regarding adaptation to chronic illness. For instance, the above results demonstrate the potential for vast variability in one's degree of adaptation over a relatively short time period of time (the time between T1 and T2 in this instance was little over a year). Furthermore, this variability was shown to manifest in both directions, with some patients demonstrating increased adaptation and others demonstrating decreased adaptation. As such, these results find reflection in the literature in that adaptation has repeatedly been advocated as being non-linear (Stanton et al., 2007). In conjunction with this, the qualitative results demonstrate the vast array of factors that contribute to one's degree of adaptation and are thus antecedents of the factors of the A-IBD. For example, the current study found that besides disease-related factors, multiple other factors play an important role in determining adaptation to IBD such as social factors, psychological resilience, and one's willingness to engage with their environment in general. This too is put forward in the adaptation literature 151 in that outside of the physical limitations that diseases impose during periods of illness exacerbation, other contextual factors such as the understanding of those in one's work environment, for example, is also important to consider. Consequently, the current study demonstrates the importance of evaluating the context of the patient in understanding their adaptation, appreciating that heterogeneity is the rule, not the exception (Larsen & Hummel, 2012). 4.5.5 In Summary Overall, the current study builds on the narrative of this thesis by, firstly, uncovering what the potential factors are that contribute to possible fluctuations in one's degree of adaptation to IBD over time. As such, not only has the current thesis conceptualised and operationalised adaptation to IBD where it was previously lacking, as well as ascertaining the utility of understanding this concept in terms of its ability to predict QoL, but we have now reached a point whereby we are understanding of the antecedents of this adaptation. In conjunction, we now understand that high adaptation as measured by the A-IBD is not merely associated with higher QoL, but the current study's findings reflect notions of self-care as well, highlighted by the subthemes of 'honesty' and 'openness'. For example, in the former, those high in adaptation were honest with themselves about when their fatigue was likely at a point where further exertion would result in harm. With regards to the latter meanwhile, those high in adaptation were also open and transparent about their illness with friends and in their work environment in case they should feel unable to socialise or manage their work. Consequently, although the current thesis has advocated adaptation for its ability in predicting QoL, it also is an appropriate process to understand since it is evident that those high in adaptation also evidence appropriate self-care. Furthermore, the current study confirms the concept of adaptation is indeed dynamic and thus susceptible to change over time. 152 With a fully developed understanding of the meaning of adaptation in terms of those with IBD, not only can better interventions now be designed around this knowledge, but the template by which this understanding was developed can now be applied to other illness populations in an attempt to conceptualise, operationalise and alter adaptation in these patients as well with the aim of improving patient QoL. 4.5.6 Methodological limitations With regards to the limitations of the current study, there are arguably some issues regarding the subsample of patients with IBD that were involved. Firstly, the participants that were recruited for this study were a subsample of the 307 participants recruited in Study 2 (Chapter 3), of which participants could be of any nationality or culture. However, the current study reverted to the recruitment criteria used in Study 1's thematic analysis whereby participants were only eligible to participate if they were living within the UK. While this was decided upon for pragmatic reasons, it does raise an inconsistency in the sampling method and bring into question whether the measure of A-IBD as it was determined in Study 2 is applicable if it is applied to an exclusively UK-based sample of patients with IBD. Furthermore, the selection criterion of those demonstrating high and low adaptation as being in the top and bottom 25% of scorers on the A-IBD from Study 2 is a relatively subjective determination of high and low adaptation, respectively. The criterion of 25% from the top and bottom scorers was decided upon to ensure a sufficiently large pool of prospective participants was available to contact and allow for the interviews to be commenced as quickly as possible. While the subsequent thematic analysis did reveal some rich data as to the differences between the high and low scorers based on this 25% criterion, it is possible that the results may have differed more extremely had the participants been from, for example, the top and bottom 10% of scorers on the A-IBD. 153 In conjunction with this, with regards to the study design, participants were asked to reflect on the answers they had given to the A-IBD from Study 2 despite haven given these answers a year prior to the current study. Due to the demonstrated dynamic nature of adaptation it is likely that participants may have struggled to recall their state of mind and reasoning behind their responses and indeed this was evident on occasion during the interviews. In addition, since the participants knew they would be interviewed regarding their A-IBD scores, it is feasible that a certain degree of desirability bias may have existed in their responses in that patients may have under-reported on aspects they deemed to be undesirable (for items such as "I fear that my illness will stop me achieving my goals in life") and overreported on seemingly desirable aspects (such as "I still keep up with my hobbies and interests that I had before my diagnosis"). 4.5.7 Conclusion To conclude, this study provides support for the notion that a number of contextual factors contribute to one's degree of adaptation to chronic illness. In this instance, adaptation to IBD is underpinned by three broad factors - one's level of engagement, one's ability to demonstrate resilience, and certain disease-related and social contingencies. Furthermore, this study demonstrates that adaptation is a dynamic concept, evidencing this within a subsample of patients with IBD. As such, adaptation is a state rather than a trait and has indeed been shown in the current study to be susceptible to change. 154 Chapter Five General Discussion 5.1 Overview Despite much research on the effect constructs such as sense making and coping on illness outcomes in IBD, little has been done to understand the ways in which those living with IBD adapt to life with the illness. In line with this, the efficacy of psychological interventions tailored to improve outcomes in this patient population remains poor. As such, this thesis therefore aimed to address this gap in the existing literature by focusing on the notion of adaptation to IBD in terms of its conceptualisation and operationalisation and its utility as a tool for predicting patient outcomes in terms of quality of life. Specifically, it first sought to conceptualise adaptation to IBD taking a bottom-up approach, interviewing patients as to the psychological, emotional, behavioural and social changes they had experienced over time with their illness. Next it aimed to operationalise this construct, to develop a new tool and to assess the uniqueness of this tool against existing measures of psychological processes. Further, this thesis aimed to assess the ability of this new tool to predict quality of life over and above existing measures of sense making. Finally, the thesis involved interviews with those patients demonstrating high and low adaptation as a means to explore the factors that either promote or inhibit adaptation. These aims were achieved through four empirical studies. This final chapter will briefly present the findings of each of these studies. It will then highlight the links our findings share with the prior literature, outline the key themes across the thesis, discuss the methodological limitations associated with the thesis and, finally, describe the implications of the findings for both research and practice. 155 5.2 Summary of findings Study 1 used a qualitative design to explore how patients with IBD experience their condition and how, if at all, they changed over time psychologically, emotionally, behaviourally and socially. A thematic analysis revealed three core themes relating to 'making sense of the illness', the 'impact' associated with the illness, and the common 'feelings' experienced by the patients in relation to their disease. Transcending these three themes was an overarching notion of 'uncertainty' which was subsequently resolved through a process employing key specific psychological, social and behavioural coping methods. Central to this process was a balance between their new identity as a patient with their existing identity as a person who manages a chronic illness. This reflected a novel way to conceptualise the notion of adaptation in patients with IBD. Study 2 employed a cross-sectional, quantitative design to operationalise adaptation to IBD into a reliable measure of the concept. Psychometric analysis of an initial 40-item questionnaire resulted in an 18-item measure of adaptation (termed the A-IBD) comprised of four subscales reflecting 'patient identity', 'person identity', 'acceptance' and 'expectations'. This measure demonstrated mostly good internal reliability based on Cronbach's alphas analysis. This measure enables patients' adaptation to IBD to be quantified in a way that reflects their experiences. Study 2 also study sought to investigate whether the A-IBD was indeed measuring something distinct to already established measures of psychological processes in the form of sense making, as measured by the BIPQ, and outcomes in the form of QoL, as measured by the IBDQ. Finally, this chapter aimed to assess the predictive ability of the A-IBD with regards to QoL as measured by the IBDQ, as well as whether the A-IBD could predict IBDQ scores over and above the BIPQ. The results suggested that the A-IBD did indeed measure a construct distinct from that measured by the BIPQ or IBDQ since correlations between the former and latter measures were weak to moderate. Further, multiple 156 regression analyses revealed that the A-IBD significantly predicted both IBDQ and IBDQ-S scores (a variation of the IBDQ specifically designed for patients with a stoma) and predicted an extra 5% and 6% of the variance in IBDQ/S scores, respectively, compared to when the BIPQ predicted IBDQ/S scores alone, justifying the utility of the A-IBD. This study highlighted the particular importance of identity in impacting QoL scores as suggested by the multiple regression model. However, differences in the regression models were evident between the non-stoma and stoma samples, suggesting further research is warranted into adaptation specific to those patients with stomas. Finally, Study 3 sought, primarily, to conduct another qualitative, interview-based analysis of the factors that the top and bottom 25% of scorers on the A-IBD from Chapter 3 felt encouraged or inhibited their degree of adaptation to their illness. The study also utilised a quantitative design to investigate changes in adaptation as measured by the A-IBD over time since study 2. In doing so, the aim was to assess whether adaptation was indeed a dynamic concept as prior literature had suggested and thus a state rather than a trait. Three emergent themes were found upon thematic analysis of the qualitative data including 'engagement', 'resilience' and 'contingencies'. While the former two themes tended to be characteristics of higher adapted participants, the contingencies subthemes of 'disease factors' and 'social factors' put forward a possible explanation as to the reason behind why some patients may demonstrate low adaptation. Furthermore, upon observation of the change in adaptation scores of the participants between studies 2 and 3, adaptation was indeed proven to be a state rather than trait, with six participants displaying no change in adaptation score while three participants improved and six deteriorated. It is suggested that with the knowledge gained in this Chapter as to the antecedents of high and low adaptation that future interventions target these factors in order to maximise patient QoL. 157 5.3 Story of the thesis - What is adaptation? This thesis has explored the notion of adaptation. A number of key issues concerning the notion of adaptation can be seen to transcend the four empirical studies relating to adaptation as a response to the crisis of illness, as the prevalence of person identity over patient identity, as partially dependent on contextual variables, as holistic and as dynamic. These will now be discussed. Adaptation as a response to the crisis of illness From interviewing patients with IBD in Study 1 (Chapter 2) it was evident that being diagnosed upset these patients' inner equilibrium. The lack of understanding of the illness and the misconceptions as to what IBD entailed, combined with the all-encompassing impacts of this newly acquired burden as well as the feelings of anxiety and animosity that these bore on the patient, ultimately culminating in uncertainty over medication side effects and uncertainty over future surgery risks and symptom relapse were, as a whole, reminiscent of Moos and Schaefer's (1984) theory on the process of coping with chronic illness. They argued that illness represents a crisis and a turning point in the life of the patient, creating disequilibrium which is reminiscent of the main underlying theme of this first Study in that at the heart of IBD, and possibly other chronic illnesses, is the sense of loss of what was the patient's life before diagnosis. Indeed, Linveh and Antonak's (2005) primer on psychosocial adaptation to chronic illness and disability discusses how aspects such as 'anger/hostility', 'uncertainty and unpredictability' and issues of 'self-concept' were hallmarks of the reactions to diagnosis that patients with IBD divulged. 158 Adaptation as the prevalence of person identity over patient identity In conceptualising adaptation to IBD as primarily the balance between one's person identity with their patient identity in Study 1 (Chapter 2), it was felt that this represented an ideal goal for the patient. It was felt that by contained within the patient identity were notions of self-care such as medication adherence, acknowledging disease-related fatigue and limitations, while the person identity encouraged the sense of self and fulfilment. However, this conceptualisation was slightly altered from Study 1 upon, firstly, the results of Study 2's multiple regression analysis on what subscales of the A-IBD predicted QoL and then confirmed by Study 3's (Chapter 4) qualitative analysis on the characteristics of those demonstrating high and low adaptation. It was found here that self-care is in fact a characteristic of the person identity rather than patient identity. As a result, our conceptualisation of adaptation to IBD is not one of balance between these two identities (along with the encouragement of acceptance and expectations as outlined in the scale development chapters of this thesis), but the prevalence of the person identity over the patient identity. Consequently, patient identity is more associated with a regression to a state that is reminiscent of the initial 'crisis' period after diagnosis alluded to earlier, while person identity represents true adaptation, encapsulating notions of the sense of self as well as crucial practical, self-care elements that are necessary for wellbeing. Adaptation is partially dependent on contextual variables Continuing the theme of adaptation as a biopsychosocial construct, often in the psychological literature there is an underlying notion that psychology alone can explain or predict an outcome, particularly evident when we consult theories of sense making such as Taylor's Cognitive Adaptation Theory (1983) or Leventhal et al.'s (1980) Self-Regulatory Model. However, one of the motivations of this thesis was to portray a slightly different 159 outlook on chronic illness and the patient at the heart of it. This was particularly evident in Study 3 (Chapter 4) where the aim of the Study was to move from what Studies 1 and 2 had achieved in understanding what adaptation to IBD is, to how does one reach that point and why do some fail to do so. In asking this question we discovered that a common issue for those demonstrating low adaptation were social and disease-related factors. Granted these participants' scores may well improve following an intervention predicated on the findings of this thesis which would hopefully improve their psychological functioning and possibly their disease status and social functioning as a by-product, but the resounding message here really is that contextual factors cannot be discredited. Indeed if we consult Linveh's (2001) conceptual framework to psychosocial adaptation, he specifically states that there are antecedents to adaptation (as was emphasised in Study 3) including contextual variables such as 'biological and biographical status' (current health status, level of physical maturity, ethnicity), 'psychosocial status' (and 'environmental conditions'. Adaptation is holistic Typically in the psychological literature it is observed that adaptation or 'adjustment' as it is often termed is observed from a more or less purely psychological standpoint. The nursing literature meanwhile tends to prefer the term 'psychosocial' adjustment or adaptation to take into account the social environment of the patient as well as their psychological state since one does not exist without the other (Larsen, 2016). Similarly, medicine tends to focus on the biomedical aspects of chronic illness. This thesis proposes that the both perspectives are incomplete. If we are to comprehend chronic illness then we must take a holistic approach as its impacts are multifaceted and span social, cognitive, emotional, physiological and behavioural domains, with all domains changing constantly. This has been shown to be true of IBD and therefore there is no reason to believe it's not true of other chronic illnesses. Research as far back as that of Vistosky et al. (1961) on patients with polio posed a simple 160 question to these patients in how it was possible to deal with the stressors of their illness and which coping behaviours assisted them in achieving an optimal outcome. These are the basic principles upon which this thesis was founded, developing an understanding on the process of adaptation as a result of understanding, first of all, the lived experience of the patient and not viewing this solely through a psychological prism as was evident with the design of Study 1 (Chapter 2). Adaptation is dynamic As adaptation is holistic, dependent on the interplay of an array of variables, adaptation is also therefore dynamic, changing incrementally on a constant basis as any one of the factors that determine it varies. Study 3 (Chapter 4) demonstrated this fact, and although a number of participants demonstrated minor changes in adaptation, it should be remembered that the design was cross-sectional in nature and thus doesn't account for the fluctuations in adaptation between the two time points where it was measured. In summary This thesis has consisted of four empirical studies which have explored the concept of adaptation in patients with IBD. From this thesis it is concluded that adaptation is a dynamic and holistic construct which is a response to the crisis of illness, related to contextual variables, and illuminates a tension between the person’s identity as a patient with IBD versus that of a person without. 5.4 Methodological limitations As is characteristic of all research, each of the studies comprising this thesis carry methodological limitations. Regarding first the recruitment approach, a number of issues can be raised. Firstly, choosing to recruit participants for this thesis solely from online sources 161 brings up issues of sample bias since the participants recruited from this source may not be representative to the wider IBD population. Consequently, issues with the degree to which all the results and conclusions in the current thesis can be interpreted and applied to, for example, regular outpatients at an IBD clinic. In other words, throughout all the studies one must consider the degree to which the deductions made are externally valid. However, this concern is somewhat alleviated by latest UK figures from the Office for National Statistics (2016) who suggested that almost all adults aged 16 to 44 had recently used the internet (98.8% to be precise), and as much as 88.3% of 55 to 64 year olds had also been online within the last 3 months. These numbers do fall for 65 to 74 year olds (74.1%) and drastically so for 75 year olds and over (38.7%), suggesting also perhaps patients from these latter age groupings were under represented in the current thesis. Indeed the oldest mean age range from any of the groups involved in this thesis was Study 3's (Chapter 4) low adaptation group where the mean age was 41.0 years. Yet despite these, for the most part, encouraging figures, it does not necessarily mean that these internet users accessed the internet for healthcare reasons and thus the concern of extrapolating our results to a general IBD populations is valid. In continuation, caution must also be taken when interpreting the findings due to one particular disparity in the inclusion criteria between the studies, revolving around the criterion of participants being required to be a UK resident in Studies 1 and 4, but not in Study 2 (Study 3 also used the sample from Study 2) whereby participants were recruited worldwide. Ideally, the criteria for the studies would have been consistent throughout, however due to the large number of participants required to sufficiently conduct in particular Study 2's factor analysis of the adaptation measure. In line with this, in this instance it was felt that it would be more pragmatic to allow for a wider participant pool. The offshoot of this decision though is the concern of whether the mode of adaptation to IBD differs between cultures. A final concern on the samples involved in this thesis revolves around the gender 162 bias that was evident throughout with female participants outnumbering their male counterparts in every Study. While this again raises concerns over the generalisability of this thesis' assertions since they could be considered gynocentric, it is important to account for the fact that not only is IBD more prevalent in females (Dahlhamer et al., 2016), albeit most of these statistics are centred on Western studies, but females in psychological research generally tend to be overrepresented (Rao & Donaldson, 2015). Furthermore, the present gender bias may require less consideration if one accounts for theories such as Hyde's (2005) 'gender similarities hypothesis' which is based on findings that suggest males and females are largely more alike than different on most psychological variables. In continuation, with regards to the A-IBD, the instructions given to the participants at the time of questionnaire completion did not specifically state that the responses given to the statements of the A-IBD should be in relation to their IBD diagnosis. This lack of instruction may have framed the way participants responded to some of the items. For example, some participants may have felt that the item “I believe my fatigue will sort itself out eventually”, was not necessarily being with regards to IBD-specific fatigue as they perceived it. Finally, a limitation in the methodology of this thesis exists in the decision to not control for disease activity of the participants throughout. This decision was made upon assessment of a multitude of studies over time that have suggested disease activity was not necessarily the best predictor of QoL in IBD (Gazzard et al., 1978; Drossman et al., 1989; Mussell et al., 2004; van der Have et al., 2014) or did not impact psychological functioning (Larsson et al., 2008) . Furthermore, there is no consensus as to the most accurate and complete measure of disease activity in IBD and even when any of the suggested metrics is applied, IBD patients often experience Irritable Bowel Syndrome (IBS) symptoms which would not impact, for example, disease activity measures that involve biomarkers or endoscopic analysis. Due to this ambiguity, it was felt that avoiding controlling for disease 163 activity was justifiable. However, there are studies that have argued that disease activity is in fact the biggest predictor of QoL (Vidal et al., 2008; Kalafateli et al., 2013), and if this is in fact the case then the results of the current thesis may have to again be interpreted with caution as this possible confounding variable may impact the thesis' internal validity. Indeed, one of the subthemes of Study 3's qualitative analysis did suggest disease factors had a role in accounting for those demonstrating low adaptation scores according to the A-IBD. 5.5 Implications for literature The constant battle to retain a sense and thus adapt, as conceptualised in this instance by the struggle between the person and patient identity, is the predominant theme that runs throughout this thesis. In particular, this metaphysical concept finds much reflection in the work of those such as Charmaz on her research into adaptation and its subsequent impact on identity change. Charmaz (1995) dictated that adapting to impairment means altering life and self to accommodate to bodily losses and limits and resolving the lost unity between body and self, and that it is also struggling with rather than against the illness. Charmaz also describes the process of adaptation as consisting of three stages: experiencing impairment, making bodily assessments and identity trade-offs as people weigh their losses and gains, and finally surrendering to the impairment by relinquishing control over the illness and by flowing with the experience of it. This summary largely reflects the conceptualisation of adaptation in this thesis as the new normal being a point where those highly adapted patients demonstrate a person identity rather than a patient identity. Furthermore, Charmaz describes this process as repeatedly occurring, as was evidenced by the results of the Study 3 (Chapter 4) adaptation comparison scores with Study 2 (Chapter 3). Other similar conceptualisations have been put forward by Morea et al. (2008) who argued that upon diagnosis of a chronic illness, the 'illness self-concept' emerges whereby the illness is integrated into the self, which in turn affects adaptation to the illness. In this case, the person and patient identity may both 164 be facets of this overarching illness self-concept, with the more predominant identity predicting either positive or negative adaptation, respectively. Interestingly, Morea et al. (2008) also found that in operationalising the illness self-concept, it predicted unique variance in the health-related outcomes of fibromyalgia patients where individual aspects such as optimism and self-esteem did not. In conjunction, our findings echo Breakwell's (1986) identity process theory, similar in both its concept but also the fact that identity is put forward as a dynamic process as this thesis also demonstrated. In this theory, Breakwell argued that identity is shaped by a process of assimilation and accommodation whereby assimilation involves the influx of new information and accommodation represents the adaptation that occurs around this new information. In this respect, this theory may find reflection in this thesis by comparing those who demonstrate high adaptation as better accommodators of IBD and the constantly evolving challenges it faces, demonstrating better malleability and thus a better ability to deal with setbacks than those who demonstrate a more pronounced patient identity. Therefore, where the current thesis builds on popular paradigms of understanding the psychological processes behind adaptation, such as Leventhal et al.'s (1980) SRM and Taylor's (1983) CAT, as well as much of the coping literature, is emphasising the notion of the individual as an emergent complex system. Psychological processes are important, but the boundary of an individual is vast and if we are to understand adaptation then we must appreciate that adaptation is also a product of interactions between the patient and their environment as put forward by Griffiths (2009) and that the individual, or the identity of the patient and subsequent adaptation as it pertains to this thesis, emerges through a "dynamic play of relationships that occur over time" (Battersby, 1998, p. 200). Models of adaptation such as that of Moss-Morris (2013) do account for adaptation as a product of multiple factors, but classic perspectives have failed to do so. 165 This is not to completely discredit these seminal theories and popular paradigms of examining illness outcomes, but rather that the process of adaptation as it is conceptualised in this thesis is a broad, overarching concept that contains specific instances of these theories. For instance, the 'expectations' finds reflection in both CAT and the SRM in that the results of Study 3's (Chapter 4) multiple regression analysis found that higher expectations were predictive of higher QoL, resembling the 'positive illusions' aspect of CAT and the notion of 'personal control' and 'treatment control' in the SRM where both were predictive of adaptive outcomes. Meanwhile, the theme of 'fulfilment' found in Study 3 (Chapter 4) as being a characteristic of highly adapted patients echoes the notion of 'active coping'. However, once again this thesis diverges from the popular method of categorising coping as 'active' or 'passive' since this thesis also found that 'honesty' regarding fatigue and understanding limitations in order to maintain wellbeing was also a characteristic of highly adapted patients and yet this would often be classified as a passive coping approach. As such, the current thesis focused on specific coping instances, taking into account the intention of the coping and whether this was adaptive, rather than associating high and low adapters with broad typologies of coping. 5.6 Implications for research Future research should first of all address some of the above methodological limitations associated with the current thesis. As such, standardising the A-IBD in other cultures would be a valuable next step in the process of validating the measure as it not only makes the measure applicable in these cultures but also would provide further evidence of construct validity. This validation in other cultures is also important for ethical reasons as if it is implemented as part of a treatment protocol with a view to improving QoL but is not effective due to being standardised mainly in the UK, then this may potentially have negative repercussions on the patient rather than positive ones in that adaptation may be poorly 166 measured and the patient may then not receive appropriate psychological attention. Future validation studies should also look to address whether explicitly instructing the patients to answer the A-IBD in response to their IBD diagnosis alters the findings at all to those found in the current thesis. In continuation, investigating the meaning of adaptation in elderly patients with IBD would be worthwhile, but perhaps even more worthwhile perhaps would be to apply the findings from this thesis in a pediatric setting. Pediatric IBD occurs at a critical period of children's' development when the transition from pediatric to adult care can be challenging (Karwowski et al., 2009), and in fact research has suggested that adolescents with IBD tend to be more depressed than youth with other chronic diseases (Engstrom, 1992). The notions of identity prevalent in this thesis are particularly applicable in this demographic since aspects of development such as brain maturation of emotional regulation, cognitive capacity, and behavioural impulse control are continuously occuring at this point (Mackner et al., 2013) and can influence a child's adaptation as a result. As such, the self-concept of the child at this time in their life is in flux and the added challenges of IBD only compound this further, so giving adolescents guidance on the best ways to retain their person identity can protect from the myriad of factors during this age that can deteriorate QoL. In addition to this, future research should look to address the discussed limitation of controlling for disease activity. Disease factors were mentioned as a possible contributing factor to those scoring low in adaptation, and as such it would be valuable to know whether disease activity moderates the relationship between adaptation scores, as mentioned by the AIBD and outcomes such as QoL, or whether disease activity is in fact a mediator of these constructs. However, research would have to devise a way of reconciling the myriad of ways by which disease activity could be measured. The current thesis used a subjective measure to assess QoL in the IBDQ and this could be one approach to measuring disease activity by 167 using the Crohn's Disease Activity Index (CDAI; Best et al., 1976) and the Ulcerative Colitis Disease Activity Index (UCDAI; Sutherland et al., 1987). However, these measures, and the CDAI in particular, have come under some scrutiny for focusing too much on criteria such as diarrhoea, abdominal pain and sense of wellbeing, without objective measures of inflammation such as mucosal healing and biomarkers of inflammation like C-reactive protein (Leake, 2013). As a result, future research will likely need to work towards a metric that satisfies factors that are subjectively important to the patient, but also satisfy clinicians, and then investigate this as a moderator or mediator of outcome measures such as QoL. While the processes of sense making and coping, for example, have been investigated in their relationship with outcomes such as QoL, the relationship between these processes and other outcomes have also been looked into such as activity impairment (van der Have et al., 2015) , psychological distress (McCorry et al., 2009) as well as sexual health and functioning (Knowles et al., 2013). While these would be interesting to evaluate in terms of their relationship with A-IBD scores, particularly in light of the new IBD-specific distress scale (The IBD-Distress Scale; Woodward et al., 2016). Of particular relevance to the IBD population though would be an assessment of the relationship between adaptation and anxiety and depression due to the high prevalence of these concomitant comorbidities in this illness group, as well as the link between these mood disorders and relapses of symptoms in patients with IBD (Bitton et al., 2008). In carrying out this research we may develop an understanding as to whether adaptation is the 'gold standard' in terms of predicting outcomes or whether it's specific only to QoL. Finally, future research to address the cross-sectional nature of this thesis' approach to conceptualising adaptation would be beneficial. Based on findings in the coping literature that specific coping strategies may be beneficial at specific points on an illness trajectory (Petrak et al., 2001; Blalock et al., 1993), it would be worthwhile to investigate the nature of 168 adaptation to IBD at different time points and accordingly develop interventions that are catered for a patient at a particular point on their disease timeline. 5.7 Implications for practice The findings from the current thesis have a number of potential implications in the treatment protocol for patients with IBD. On an individual level, it would be worthwhile to incorporate direct, regular assessments by a psychologist or team of psychologists that are part of a wider, multifaceted healthcare team. Currently, for the most part, psychologists are either not involved at all in the treatment of those with IBD or only involved in a select few hospitals. This thesis however provides support for the aforementioned research on determinants of patient QoL that suggests the notion that treatment of IBD should be holistic since disease activity has not consistently been shown over time to be unequivocally the biggest concern for patients. Furthermore, this thesis has highlighted the dynamic interactions that occur between the patient and their illness, with both the patient and the illness affecting one another as two separate entities. As such, although, for example, we may not consider disease activity to be the most significant predictor of QoL in all instances, it does likely have a degree of impact which contributes to lowering patient QoL, which then in turn likely has an impact on the identity factors found in this thesis, encouraging patient identity and inhibiting person identity as was alluded to in the Study 3 themes when disease factors were found to be a common trait of those low in adaptation. Of course much of the onus here is on the consultants and other members of the medical team to ensure that disease activity is controlled through the administration of an appropriate treatment regime, but also there is a role here for psychologists to make patients aware of the factors they can potentially take control of in spite of their disease activity which have been shown here to predict improved QoL. Apart from perhaps the most extreme of cases, disease activity does not preclude one from engendering a person identity. Psychologists can encourage patient empowerment by 169 making them more aware of the factors that contribute to this identity as shown in Study 3 encouraging them to engage with their environment and personal passions, devising methods of improving resilience which is a buffer to illness that can be acquired at any stage of life, irrespective of age and disease status (Ghanei Gheshlagh et al., 2016). Consequently, even in times of enhanced disease activity, the intervention of psychologists can help break this negative feedback loop. Indeed on a more general level, this thesis should hopefully provide the framework for more effective psychological interventions for IBD to be formulated. Systematic reviews have suggested there is no evidence for the long-term efficacy of psychological therapy in adult patients with IBD in general, indicating that while some approaches such as psychotherapy or education-based interventions may have a short term effects on QoL and emotional status, typically at around 12 months these effects dissipate (Timmer et al., 2011). With the insight gained from this thesis it is hope that we may be able to lengthen the effects of these interventions by using frameworks that have shown promise in improving outcomes in IBD such as CBT (Mikocka-Walus et al. 2015) and mindfulness-based cognitive therapy (Berrill et al., 2014) but adapting these frameworks in line with the critical factors shown in this thesis to be related to the process of adaptation and the outcome of improved QoL. Indeed, multicentre RCTs conducted by the likes of van den Brink et al. (2016) on diseasespecific CBT is already paving the way for this approach. 5.8 Conclusion To date there has been extensive research already conducted into the psychological correlates of illness outcomes in IBD, predicated largely on popular models and perspectives of key psychological constricts. However, the notion of how these concepts come together to form the overarching process of adaptation to IBD, as well as numerous other chronic 170 illnesses, and whether this concept of adaptation has utility in predicting illness outcomes has received very little attention. Through the four empirical research studies that comprise this thesis some progress has been made towards understanding this concept, approaching the task of adaptation from the bottom-up, focusing holistically on the patient experience at the heart of the process. The result of this endeavour is a new psychometrically robust measure to assess adaptation which reflects a combination of patient identity, person identity, expectations and acceptance which has been shown to relate to patient health outcomes. 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Acta Psychiatrica Scandinavia, 67, 361-370. 195 Appendices Appendix A - Study One Information Sheet Participant Information Sheet An exploration into how Inflammatory Bowel Disease patients adapt to their illness Researchers Lawrence Matini, PhD Student, University of Surrey, Guildford, UK Professor Jane Ogden, Professor of Psychology, University of Surrey, Guildford, UK Introduction I am a first year PhD student inviting you to take part in a research study. Before you decide you want to participate it is important for you to understand why the research is being done and what it will involve. Please take the time to read the following information carefully and discuss it family or friends if you wish. If you would like any more information, feel free to contact the primary researcher (Lawrence Matini, [email protected]). Thank you for reading this. What is the purpose of the study? 196 This study seeks to gain an understanding of how patients with Inflammatory Bowel Disease adapt to their illness. In order to do this we are looking to explore the ways in which these patients think, feel and behave with regards to their illness and whether any of these thoughts, feelings or behaviours change over time. Do I have to take part? No, you do not have to participate. It is up to you to decide whether or not to take part. If you do decide that you wish to take part you will be asked to complete a short form to indicate your consent. If you decide to participate you can still change your mind and withdraw from the study entirely at any time by simply informing the researcher that you wish to do so. There will be absolutely no consequences should you wish to withdraw and your data will be deleted immediately. What will happen to me if I take part? You will be asked to take part in a 30-45 minute telephone interview during which you will be asked questions about your history with IBD, and whether any thoughts, feelings or behaviours associated with the illness have changed over time. What are the possible disadvantages or risks of taking part? We do not anticipate any serious risks of taking part. Although discussing one’s illness has the potential to be emotional and evoke upsetting memories, as mentioned earlier you are within your right to take a break from the interview or withdraw entirely from the study if you are too uncomfortable or upset to continue. What are the possible benefits of taking part? A number of benefits would be offered for your participation. Your participation will help to expand this area of research and help to further understanding of patients’ experiences of living with Inflammatory Bowel Disease. You will also contribute to the furthering of psychological interventions aimed at improving the wellbeing of IBD patients, and provide 197 information that will specifically aid in the understanding of adaptation to IBD and potentially other chronic illnesses alike. What happens when the research study stops? This study will contribute to part of my PhD. However, I also hope to publish the results of this study in a scientific journal. I may also present the results at a scientific conference or a university seminar. I would be happy to discuss the results of the study with you and to send you a copy of the published results once they are available. It will not be possible to identify any information you should provide in any report or publication as you will be given a pseudonym. What if there is a problem? Any complaint or concern about any aspect of the study or the way in which you have been treated during the course of the study will be addressed; please contact either Lawrence Matini, [email protected] or Professor Jane Ogden, [email protected] Will my taking part in the study be kept confidential? Yes. The information you provide during the study will be kept strictly confidential in any written work. We will need to analyse and store all the results. Only the principal investigator will have access to this stored material. Data will be stored securely for 10 years and then destroyed in accordance with the UK Data Protection Act (1998). Contact details of researcher and, where appropriate supervisor? Principal Investigator: Lawrence Matini, email: [email protected] Supervisor: Professor Jane Ogden, Professor in Psychology and Director of the Psychology PhD programme, University of Surrey, Guildford, UK 198 Who is organising and funding the research? This research is organised by Lawrence Matini as part of a PhD project under the supervision of Professor Jane Ogden. Who has reviewed the project? The study has been reviewed and received a favourable opinion from the University of Surrey Ethics Committee. Reference number: EC/2014/07/FAHS Thank you for taking the time to read this Information Sheet. If you would like to take part in this study, please contact Lawrence Matini to arrange a time for an interview. 199 Appendix B - Study One Consent Form Participant Consent Form I the undersigned voluntarily agree to take part in the study on the experiences and adaptation to illness in Inflammatory Bowel Disease. I have read and understood the Information Sheet provided. I have been given a full explanation by the investigators of the nature, purpose, location and likely duration of the study, and of what I will be expected to do. I have been advised about any discomfort and possible ill-effects on my health and well-being which may result. I also understand and consent to the telephone interview being recorded. I understand that I am free to withdraw from the study at any time without needing to justify my decision and without prejudice. I confirm that I have read and understood the above and freely consent to participating in this study. I have been given adequate time to consider my participation and agree to comply with the instructions and restrictions of the study. Full name: Date: 200 Appendix C - Study One Interview Schedule Hi, my name is Lawrence Matini and I am carrying out some research on Inflammatory Bowel Disease that will be included in the first study of my PhD thesis. Volunteering your time to take part is greatly appreciated and I hope you find the experience a pleasant one. I would like to ask some questions first about your history with IBD, then move on to how your life was in the early days of your diagnosis, and end with how, if at all, your feelings and behaviours have changed over time with regards to your illness. I hope to use the responses you provide to develop a better understanding of the experiences of IBD patients and how patients like yourself adapt to your illness. The interview should take about 30 minutes and will be recorded. Unless you have any questions then I am ready to begin when you are. 1) Can you tell me when you were first diagnosed with IBD? 2) How did this diagnosis come about? (transition to the next phase of questions) 3) What were your initial thoughts and feelings about your diagnosis? 4) What were your coping strategies, if any, in the early stages of your illness? (transition to the final phase of questions) 5) How do you currently feel about your diagnosis of IBD? 6) What factors have helped you manage your illness? 7) What are the biggest changes you have had to make to your life to better deal with your illness? Emotionally, socially, behaviourally… That is all the questions I have for you. I would quickly like to reiterate my appreciation for your participation and offer you the chance to ask any questions about any aspect of your involvement in the study. Should I have any further questions would it be OK to call you on this number? Thank you once again. 201 Appendix D - Study One Debrief Sheet Debrief Sheet The development of a new quantitative measure of adaptation to Inflammatory Bowel Disease and an evaluation of the role of adaptation in predicting quality of life You have now come to the end of the study. We would like to thank you for your participation and provide you with some information about the study you just took part in. Psychological research has provided much in terms of advancing the understanding of IBD and improving its prognosis. For example, research shows that whilst stress can make the disease worse, factors such as coping and social support can lead to improvements. To date, however, little is known about how psychological, social and behavioural factors combine to form adaptation to an illness, in this case IBD, and whether this notion can be measured. Further to this, it is yet to be found whether a measure of adaptation can predict patients' quality of life. This is the focus of the current study. This study aims to build on a previous study that developed an understanding of the aspects that constitute adaptation in IBD in order to form a measure of adaptation and specifically assess whether patients who demonstrate a higher degree of adaptation experience a greater health-related quality of life than those who are less well adapted to their illness. Overall we will be looking for at least 200 people with IBD to complete this questionnaire and are hoping that we will be able to develop an accurate measurement of adaptation as a means of evaluating the effectiveness of interventions designed to improve outcomes for patients with IBD. Also, we anticipate the development of this measure will contribute to an improved awareness of IBD for healthcare practitioners and lead to improved, more focused interventions that will benefit health-related quality of life in this population. This study has been reviewed and received favourable ethical approval from the University of Surrey Ethics Committee. Reference number: EC/2014/07/FAHS. If you have 202 any concerns or would like to discuss this study further, feel free to email the principal investigator: Lawrence Matini, [email protected] Principal Investigator Professor Jane Ogden, [email protected] Supervisor Thank you for taking part. 203 Appendix E - Study One University Research Committee Approval 204 Appendix F - Information Sheet (Studies Two and Three) Participant Information Sheet An exploration into how Inflammatory Bowel Disease patients adapt to their illness Researchers Lawrence Matini, PhD Student, University of Surrey, Guildford, UK Professor Jane Ogden, Professor of Psychology, University of Surrey, Guildford, UK Introduction I am a second year PhD student inviting you to take part in a research study. Before you decide you want to participate it is important for you to understand why the research is being done and what it will involve. If you would like any more information, feel free to contact me: Lawrence Matini (primary researcher), [email protected] What is the purpose of the study? This study seeks to, firstly, develop a measure of adaptation to illness in patients with Inflammatory Bowel Disease and investigate whether those who show greater levels of adaptation experience a better quality of life compared to those who score lower on our measure of adaptation. In order to do this we have developed a questionnaire that assesses the way patients with IBD perceive and behave with regards to their illness and will look to see if these scores predict patients' subsequent quality of life scores. A small number of those scoring at the top and bottom of our measure of adaptation will then be contacted for an 205 interview on the aspects that they feel contributed to their score. This will enable us to better ways to encourage adaptation in patients with IBD. Do I have to take part? No, you do not have to participate. It is up to you to decide whether or not to take part. If you do decide that you wish to take part you will be asked to complete a short form to provide consent. If you decide to participate you can still change your mind and withdraw from the study entirely at any time by simply closing the questionnaire window. There will be absolutely no consequences should you wish to withdraw and your data will be deleted. However, data can only be deleted before your questionnaire responses have been submitted as all data is stored anonymously and thus cannot be identified for deletion. Similarly, those contacted by e-mail for an interview after the questionnaire part of the study can decline to do so without absolutely any consequences. Those who do take part in the interviews may also withdraw from that section of the study and will have their data deleted immediately. What will happen to me if I take part? You will be asked to take part in an 15-30 minute online questionnaire during which you will be asked questions about how you perceive your life with IBD and how you behave with regards to your illness which is aimed at measuring how adapted you are to your illness. You will also be asked about your current quality of life while living with IBD. A selection of those scoring at the top and bottom of the adaptation questionnaire will then be contacted to request an interview on the responses they gave in the questionnaire. What are the possible disadvantages or risks of taking part? We do not anticipate any serious risks of taking part. Although reflecting on one's illness has the potential to be emotional and evoke upsetting memories, you are within your right to take a break from the questionnaire or interview, or to even withdraw entirely from the study at any point as mentioned earlier if you are too uncomfortable or upset to continue. 206 What are the possible benefits of taking part? A number of benefits are expected from participating. Your participation will help significantly to expand this area of research and fill a large gap in the existing literature on adaptation to IBD as well as potentially other chronic illnesses. You will also hopefully contribute to the development of treatment protocols to include psychological interventions that are tailored to individual patients with the aim of improving patient wellbeing. What happens when the research study stops? This study will form a major part of my PhD. However, I also aim to publish the results of this study in a scientific journal. I may also present the results at a scientific conference or a university seminar. I would be happy to discuss the results of the study with you and to send you a copy of the published results once they are available. If this interests you, you will have an opportunity to enter your email address after the study debriefing which follows the questionnaire. What if there is a problem? Any complaint or concern about any aspect of the study will be addressed; please contact Lawrence Matini, [email protected]. Will my taking part in the study be kept confidential? Yes. The information you provide during the study will be kept strictly confidential and you will not be associated with your data in any written work. We will need to analyse and store all the results and only the principal investigator and supervisor, Professor Jane Ogden, will have access to this. Personal data will be stored securely in accordance with the UK Data Protection Act (1998). Research data will be retained securely for a minimum of 10 years. 207 Contact details of researcher and, where appropriate, supervisor? Principle Investigator: Lawrence Matini, email: [email protected] Supervisor: Professor Jane Ogden, Professor in Psychology and Director of the Psychology PhD programme, University of Surrey, Guildford, UK. email: [email protected] Head of School of Psychology: Professor Peter Hegarty. email: [email protected] Who is organising and funding the research? This research is organised by Lawrence Matini as part of a PhD project under the supervision of Professor Jane Ogden and is self funded. Who has reviewed the project? The study has been reviewed and received a favourable opinion from the University of Surrey Ethics Committee. Reference number: EC/2014/157/FAHS. Thank you for taking the time to read this Information Sheet. If you would like to take part in this study then please continue. 208 Appendix G - Consent Form (Studies Two and Three) Participant Consent Form I the undersigned voluntarily agree to take part in this study on adaptation to illness and quality of life in Inflammatory Bowel Disease. I have read and understood the Information Sheet provided. I have been given a full explanation by the investigators of the nature, purpose, location and likely duration of the study, and of what I will be expected to do. I have been advised about any discomfort and possible ill-effects on my health and well-being which may result. I also understand and consent to a telephone interview being recorded should I be selected for participation. I understand that I am free to withdraw from the study at any time without needing to justify my decision and without prejudice. I confirm that I have read and understood the above and freely consent to participating in this study. I have been given adequate time to consider my participation and agree to comply with the instructions and restrictions of the study. I consent to my personal data, as outlined in the accompanying information sheet, being used for this study. I understand that all personal data relating to participants is held and processed in the strictest confidence, and in accordance with the Data Protection Act (1998). (Participants will click a button at the bottom of this form to confirm their consent) 209 Appendix H - Debrief Sheet (Study Two) Debrief Sheet The development of a new quantitative measure of adaptation to Inflammatory Bowel Disease and an evaluation of the role of adaptation in predicting quality of life We would like to thank you for your participation and provide you with some information about the research you just took part in. Psychological research has provided much in terms of advancing the understanding of IBD and improving its prognosis. For example, research shows that while stress can make the disease worse, factors such as coping and social support can lead to improvements. To date, however, little is known about how psychological, social and behavioural factors combine to form adaptation to an illness, in this case IBD, and whether this notion can be measured. Further to this, it is yet to be found whether a measure of adaptation can predict patients' quality of life. This is the focus of the current study. This study aims to build on a previous study that developed an understanding of the aspects that constitute adaptation in IBD in order to form a measure of adaptation and specifically assess whether patients who demonstrate a higher degree of adaptation experience a greater health-related quality of life than those who are less well adapted to their illness. Overall we will be looking for at least 200 people with IBD to complete this questionnaire and are hoping that we will be able to develop an accurate measurement of adaptation as a means of evaluating the effectiveness of interventions designed to improve outcomes for patients with IBD. We anticipate the development of this measure and the subsequent interviews on the factors that contribute to adaptation will lead to an improved awareness of IBD for healthcare practitioners and lead to improved, more focused interventions that will benefit health-related quality of life in this population. 210 This study has been reviewed and received favourable ethical approval from the University of Surrey Ethics Committee. If you have any concerns or would like to discuss this study further, feel free to email the lead researcher or research supervisor: Lawrence Matini, [email protected] Principal Investigator Professor Jane Ogden, [email protected] Supervisor Please enter your e-mail address below if you would like to receive a summary of the findings and would consider taking part in a possible interview regarding your results: Thank you for taking part. 211 Appendix I - Adaptation to IBD Questionnaire (Study Two) Read the statements on the left and mark your level of agreement on the right. If you do not have an opinion on a particular statement, please mark "Neither agree nor disagree". If at any point you wish to withdraw from the study you may close the questionnaire window and your data will be deleted. Once you submit your answers your data may not be removed. Person identity 1) I forget that I have IBD (Strongly Disagree, Disagree, Neither agree nor disagree, Agree, Strongly Agree) 2) I go out and socialise regardless of any symptoms I may be experiencing 3) I experience minimal disruptions to my school or work life despite my illness 4) I still keep up with my hobbies and interests that I had before my diagnosis 5) I exert myself just as much as I used to before my diagnosis 6) I try and live a normal life like everyone else 7) I ignore any fatigue I may be experiencing and try to function as normal 8) I do not believe my illness is that serious 9) I am dismissive of my symptoms 10) My illness will not get the better of me Patient identity 1) I'm conscious that when I go out with my friends I may suddenly need the toilet 2) I think about how my illness has affected me in the past 3) I fear that my illness will stop me achieving my goals in life 4) I wonder about how my health will be in the future 5) Life will never be the same as it was before my diagnosis 6) There is nothing I can do to make myself better 7) If I exercise then my symptoms will return 8) I spend time researching medications or other treatment options 212 9) I label myself as a person with a chronic illness 10) I feel that others see me as an ill person Acceptance 1)Some days I will not be as productive as I want to be 2) I accept that I have a chronic illness which has no cure 3) I am accepting of the possibility of future flare ups of my symptoms 4) I feel just as positive about life despite my illness 5) I get angry or upset when I experience symptoms 6) There will be fluctuations in my mood with regards to my illness 7) I am comfortable discussing my symptoms with my family and close friends 8) I just have to keep moving forward with my life 9) I feel like my illness is a burden 10) I am comfortable with my body image Expectations 1) If my symptoms are under control then they will not return providing I continue my treatment 2) I will live the life I had before being diagnosed 3) I will be cured from my illness one day 4) I will achieve everything I always set out to achieve in life 5) I expect things will only get easier with time 6) I expect my doctor will eventually find a medication that solves my problems 7) Surgery will completely turn things around for me 8) I believe my fatigue will sort itself out eventually 9) I expect I will relapse at some point 10) My illness will not interfere in any way with any romantic relationships 213 Appendix J - Brief Illness Perceptions Questionnaire (Study Two) For the following questions, please circle the number that best corresponds to your views: How much does your illness affect your life? 0 – no affect at all, 1, 2, 3, 4, 5, 6, 7, 8, 9, 10 – severely affects my life How long do you think your illness will continue? 0 – a very short time, 1, 2, 3, 4, 5, 6, 7, 8, 9, 10 – forever How much control do you feel you have over your illness? 0 – absolutely no control, 1, 2, 3, 4, 5, 6, 7, 8, 9, 10 – extreme amount of control How much do you think your treatment can help your illness? 0 – not at all, 1, 2, 3, 4, 5, 6, 7, 8, 9, 10 – extremely helpful How much do you experience symptoms from your illness? 0 – no symptoms at all, 1, 2, 3, 4, 5, 6, 7, 8, 9, 10 – many severe symptoms How concerned are you about your illness? 0 – not at all concerned, 1, 2, 3, 4, 5, 6, 7, 8, 9, 10 – extremely concerned How well do you feel you understand your illness? 0 – don’t understand at all, 1, 2, 3, 4, 5, 6, 7, 8, 9, 10 – understand very clearly How much does your illness affect your emotionality? (e.g. does it make you angry, scared, upset or depressed?) 0 – not at all affected emotionally, 1, 2, 3, 4, 5, 6, 7, 8, 9, 10 – extremely affected emotionally 214 Appendix K - Inflammatory Bowel Disease Questionnaire (Study Two) This questionnaire is designed to find out how you have been feeling during the last 2 weeks. You will be asked about symptoms you have been having as a result of your inflammatory bowel disease, the way you have been feeling in general, and how your mood has been. How frequent have your bowel movements been during the last two weeks? Please indicate how frequent your bowel movements have been during the last two weeks by picking one of the options from 1 BOWEL MOVEMENTS AS OR MORE FREQUENT THAN THEY HAVE EVER BEEN 2 EXTREMELY FREQUENT 3 VERY FREQUENT 4 MODERATE INCREASE IN FREQUENCY OF BOWEL MOVEMENTS 5 SOME INCREASE IN FREQUENCY OF BOWEL MOVEMENTS 6 SLIGHT INCREASE IN FREQUENCY OF BOWEL MOVEMENTS 7 NORMAL, NO INCREASE IN FREQUENCY OF BOWEL MOVEMENTS How often has the feeling of fatigue or of being tired and worn out been a problem for you during the last 2 weeks? Please indicate how often the feeling of fatigue or tiredness has been a problem for you during the last 2 weeks by picking one of the options from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you felt frustrated, impatient, or restless? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you been unable to attend school or do your work because of your bowel problem? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How much of the time during the last 2 weeks have your bowel movements been loose? Please choose an option from 215 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How much energy have you had during the last 2 weeks? Please choose an option from 1 NO ENERGY AT ALL 2 VERY LITTLE ENERGY 3 A LITTLE ENERGY 4 SOME ENERGY 5 A MODERATE AMOUNT OF ENERGY 6 A LOT OF ENERGY 7 FULL OF ENERGY How often during the last 2 weeks did you feel worried about the possibility of needing to have surgery because of your bowel problem? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you had to delay or cancel a social engagement because of your bowel problem? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME 1. How often during the last 2 weeks have you been troubled by cramps in your abdomen? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you felt generally unwell? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 216 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you been troubled because of fear of not finding a washroom? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How much difficulty have you had, as a result of your bowel problems, doing leisure or sports activities you would have liked to have done during the last 2 weeks? Please choose an option from 1 A GREAT DEAL OF DIFFICULTY; ACTIVITIES MADE IMPOSSIBLE 2 A LOT OF DIFFICULTY 3 A FAIR BIT OF DIFFICULTY 4 SOME DIFFICULTY 5 A LITTLE DIFFICULTY 6 HARDLY ANY DIFFICULTY 7 NO DIFFICULTY; THE BOWEL PROBLEMS DID NOT LIMIT SPORTS OR LEISURE ACTIVITIES How often during the last 2 weeks have you been troubled by pain in the abdomen? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you had problems getting a good night's sleep, or been troubled by waking up during the night? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you felt depressed or discouraged? Please choose an option from 217 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you had to avoid attending events where there was no washroom close at hand? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME Overall, in the last 2 weeks, how much of a problem have you had with passing large amounts of gas? Please choose an option from 1 A MAJOR PROBLEM 2 A BIG PROBLEM 3 A SIGNIFICANT PROBLEM 4 SOME TROUBLE 5 A LITTLE TROUBLE 6 HARDLY ANY TROUBLE 7 NO TROUBLE Overall, in the last 2 weeks, how much of a problem have you had maintaining or getting to, the weight you would like to be at? Please choose an option from 1 A MAJOR PROBLEM 2 A BIG PROBLEM 3 A SIGNIFICANT PROBLEM 4 SOME TROUBLE 5 A LITTLE TROUBLE 6 HARDLY ANY TROUBLE 7 NO TROUBLE Many patients with bowel problems often have worries and anxieties related to their illness. These include worries about getting cancer, worries about never feeling any better, and worries about having a relapse. In general, how often during the last 2 weeks have you felt worried or anxious? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME 218 How much of the time during the last 2 weeks have you been troubled by a feeling of abdominal bloating? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you felt relaxed and free of tension? Please choose an option from 1 NONE OF THE TIME 2 A LITTLE OF THE TIME 3 SOME OF THE TIME 4 A GOOD BIT OF THE TIME 5 MOST OF THE TIME 6 ALMOST ALL OF THE TIME 7 ALL OF THE TIME How much of the time during the last 2 weeks have you had a problem with rectal bleeding with your bowel movements? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How much of the time during the last 2 weeks have you felt embarrassed as a result of your bowel problem? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME 1. How much of the time during the last 2 weeks have you been troubled by a feeling of having to go to the bathroom even though your bowels were empty? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How much of the time during the last 2 weeks have you felt tearful or upset? Please choose an option from 219 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How much of the time during the last 2 weeks have you been troubled by accidental soiling of your underpants? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How much of the time during the last 2 weeks have you felt angry as a result of your bowel problem? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME To what extent has your bowel problem limited sexual activity during the last 2 weeks? Please choose an option from 1 NO SEX AS A RESULT OF BOWEL DISEASE 2 MAJOR LIMITATION AS A RESULT OF BOWEL DISEASE 3 MODERATE LIMITATION AS A RESULT OF BOWEL DISEASE 4 SOME LIMITATION AS A RESULT OF BOWEL DISEASE 5 A LITTLE LIMITATION AS A RESULT OF BOWEL DISEASE 6 HARDLY ANY LIMITATION AS A RESULT OF BOWEL DISEASE 7 NO LIMITATION AS A RESULT OF BOWEL DISEASE How much of the time during the last 2 weeks have you been troubled by nausea or feeling sick to your stomach? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How much of the time during the last 2 weeks have you felt irritable? Please choose an option from 220 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the past 2 weeks have you felt a lack of understanding from others? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How satisfied, happy, or pleased have you been with your personal life during the past 2 weeks? Please choose one of the following options from 1 VERY DISSATISFIED, UNHAPPY MOST OF THE TIME 2 GENERALLY DISSATISFIED, UNHAPPY 3 SOMEWHAT DISSATISFIED, UNHAPPY 4 GENERALLY SATISFIED, PLEASED 5 SATISFIED MOST OF THE TIME, HAPPY 6 VERY SATISFIED MOST OF THE TIME, HAPPY 7 EXTREMELY SATISFIED, COULD NOT HAVE BEEN MORE HAPPY OR PLEASED 221 Appendix L - Inflammatory Bowel Disease Questionnaire For Patients With Stomas (Study Two) This questionnaire is designed to find out how you have been feeling during the last 2 weeks. You will be asked about symptoms you have been having as a result of your ulcerative colitis/Crohn’s disease or indeterminate colitis, the way you have been feeling in general, and how your mood has been. How frequently have you had to empty your colostomy or ileostomy appliance during the last two weeks? Please indicate how frequent your stomal output has been during the last two weeks by picking one of the options from 1 AS OR MORE FREQUENT THAN EVER 2 EXTREMELY FREQUENT 3 VERY FREQUENT 4 MODERATE INCREASE IN FREQUENCY OF EMPTYING 5 SOME INCREASE IN FREQUENCY OF EMPTYING 6 SLIGHT INCREASE IN FREQUENCY OF EMPTYING 7 NORMAL, NO INCREASE IN FREQUENCY OF EMPTYING How often has the feeling of fatigue or of being tired and worn out been a problem for you during the last 2 weeks? Please indicate how often the feeling of fatigue or tiredness has been a problem for you during the last 2 weeks by picking one of the options from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME 222 How often during the last 2 weeks have you felt frustrated, impatient, or restless? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you been unable to attend school or do your work because of your bowel problem? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How much of the time during the last 2 weeks has your stomal output been looser than normal? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME 223 How much energy have you had during the last 2 weeks? Please choose an option from 1 NO ENERGY AT ALL 2 VERY LITTLE ENERGY 3 A LITTLE ENERGY 4 SOME ENERGY 5 A MODERATE AMOUNT OF ENERGY 6 A LOT OF ENERGY 7 FULL OF ENERGY How often during the last 2 weeks did you feel worded about the possibility of needing to have surgery because of your bowel problem? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you had to delay or cancel a social engagement because of your bowel problem? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME 224 How often during the last 2 weeks have you been troubled by cramps in your abdomen? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you felt generally unwell? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you been troubled because of fear of not finding a washroom (bathroom, toilet)? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME 225 How much difficulty have you had, as a result of your bowel problems, doing leisure or sports activities you would have liked to have done during the last 2 weeks? Please choose an option from 1 A GREAT DEAL OF DIFFICULTY; ACTIVITIES MADE IMPOSSIBLE 2 A LOT OF DIFFICULTY 3 A FAIR BIT OF DIFFICULTY 4 SOME DIFFICULTY 5 A LITTLE DIFFICULTY 6 HARDLY ANY DIFFICULTY 7 NO DIFFICULTY; THE BOWEL PROBLEMS DID NOT LIMIT SPORTS OR LEISURE ACTIVITIES How often during the last 2 weeks have you been troubled by pain in the abdomen? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you had problems getting a good night's sleep, or been troubled by waking up during the night? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME 226 How often during the last 2 weeks have you felt depressed or discouraged? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the last 2 weeks have you had to avoid attending events where there was no washroom (bathroom, toilet) close at hand? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME Overall, in the last 2 weeks, how much of a problem have you had with your stomal appliance filling up with a large amounts of gas? Please choose an option from 1 A MAJOR PROBLEM 2 A BIG PROBLEM 3 A SIGNIFICANT PROBLEM 4 SOME TROUBLE 5 A LITTLE TROUBLE 6 HARDLY ANY TROUBLE 7 NO TROUBLE 227 Overall, in the last 2 weeks, how much of a problem have you had maintaining or getting to, the weight you would like to be at? Please choose an option from 1 A MAJOR PROBLEM 2 A BIG PROBLEM 3 A SIGNIFICANT PROBLEM 4 SOME TROUBLE 5 A LITTLE TROUBLE 6 HARDLY ANY TROUBLE 7 NO TROUBLE Many patients with bowel problems often have worries and anxieties related to their illness. These include worries about getting cancer, worries about never feeling any better, and worries about having a relapse. In general, how often during the last 2 weeks have you felt worried or anxious? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How much of the time during the last 2 weeks have you been troubled by a feeling of abdominal bloating? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME 228 How often during the last 2 weeks have you felt relaxed and free of tension? Please choose an option from 1 NONE OF THE TIME 2 A LITTLE OF THE TIME 3 SOME OF THE TIME 4 A GOOD BIT OF THE TIME 5 MOST OF THE TIME 6 ALMOST ALL OF THE TIME 7 ALL OF THE TIME How much of the time during the last 2 weeks have you had a problem with blood in your stomal output or blood from the rectum? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How much of the time during the last 2 weeks have you felt embarrassed as a result of your bowel problem? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME 229 How much of the time during the last 2 weeks have you been troubled by a feeling of having to go to the bathroom to empty your rectum, even though you have a stoma? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How much of the time during the last 2 weeks have you felt tearful or upset? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How much of the time during the last 2 weeks have you been troubled by accidental soiling of your clothing or bedding because of leaking from your stomal appliance? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME 230 How much of the time during the last 2 weeks have you felt angry as a result of your bowel problem? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME To what extent has your bowel problem limited sexual activity during the last 2 weeks? Please choose an option from 1 NO SEX AS A RESULT OF BOWEL DISEASE 2 MAJOR LIMITATION AS A RESULT OF BOWEL DISEASE 3 MODERATE LIMITATION AS A RESULT OF BOWEL DISEASE 4 SOME LIMITATION AS A RESULT OF BOWEL DISEASE 5 A LITTLE LIMITATION AS A RESULT OF BOWEL DISEASE 6 HARDLY ANY LIMITATION AS A RESULT OF BOWEL DISEASE 7 NO LIMITATION AS A RESULT OF BOWEL DISEASE How much of the time during the last 2 weeks have you been troubled by nausea or feeling sick to your stomach? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME 231 How much of the time during the last 2 weeks have you felt irritable? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How often during the past 2 weeks have you felt a lack of understanding from others? Please choose an option from 1 ALL OF THE TIME 2 MOST OF THE TIME 3 A GOOD BIT OF THE TIME 4 SOME OF THE TIME 5 A LITTLE OF THE TIME 6 HARDLY ANY OF THE TIME 7 NONE OF THE TIME How satisfied, happy, or pleased have you been with your personal life during past 2 weeks? Please choose one of the following options from 1 VERY DISSATISFIED, UNHAPPY MOST OF THE TIME 2 GENERALLY DISSATISFIED, UNHAPPY 3 SOMEWHAT DISSATISFIED, UNHAPPY 4 GENERALLY SATISFIED, PLEASED 5 SATISFIED MOST OF THE TIME, HAPPY 6 VERY SATISFIED MOST OF THE TIME, HAPPY 7 EXTREMELY SATISFIED, COULD NOT HAVE BEEN MORE HAPPY OR PLEASED 232 Appendix M - Study Three Interview Schedule Now that we have a good understanding of what adaptation involves, I would like to understand what the key aspects were that enabled patients who scored the highest on my adaptation questionnaire to do so. Likewise, I hope to understand what prevented the lower scorers from scoring more highly. The interview should take about 30 minutes and will be recorded. Unless you have any questions then I am ready to begin when you are. Establish rapport with participant 1) Can you tell me a bit about your history with IBD? (When were you diagnosed? Were you quickly diagnosed or was it a lengthy process?) 2) In your opinion what have been the major difficulties you’ve faced with respect to your illness in terms of dealing with it emotionally? Psychologically? Behaviourally? Transition to questionnaire results 3) Based on your scores from the questionnaire study you took part in, you appeared to score particularly high/low on questions relating to what I call the ‘patient identity’. These questions were to do with aspects such as fear of the future with regards to your illness and difficulties in dealing with the illness emotionally and psychologically (Refer to specific questions from patient identity section and participants’ scores for particularly low/high scoring questions). What do you feel are the main things that have prevented you from scoring lower on this scale/helped you score low on this scale? Do you feel the reasons for you score are more internal (rumination, psychological resilience) or external (support from family, healthcare team)? 4) Again, looking at your scores, you appeared to score particularly high/low on questions relating to what I call the ‘person identity’. These questions were to do with trying to live as normal a life as possible by doing things such as still keeping up with your hobbies and your friends. 233 What, in your opinion, are the main things that have helped you reach this point/prevented you from scoring higher? How do you prevent yourself from letting this mind set slip? How do you manage your health while trying to live ‘normally’? 5) The third aspect I want to look into are the scores you gave in relation to the theme of hope in my questionnaire. These questions referred to your opinion on aspects such as whether you believed your doctor would ever find a medication that solves your problems or even whether you believe you may be cured one day. Have you always felt this way? If not, then how do you think you developed this sense of hope? If you don’t feel hopeful, what is preventing you from doing so? Do past experiences shape your hope in relation to IBD? Fear of being wrong about cure? 6) And finally, I’d just like to touch on the score for the final theme of my questionnaire and that is acceptance. These questions referred to your opinion on aspects such as accepting that there is no cure for IBD and that you just have to keep moving forward with your life despite your illness. How do you feel you have developed this sense of acceptance? Does this acceptance of your illness waver and, if so, under what circumstances? Why do you feel you cannot fully accept your illness (low scorers)? That’s all the questions I have for you. I would quickly like to reiterate my appreciation for your participation and offer you the chance to ask any questions about any part of your involvement in the study. Should I have any further questions would it be OK to call you on this number? Thank you once again. 234 Appendix N - University Ethics Committee Approval (Studies Two and Three) 235