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The Effects of Operant Behavioral Management of Movement Patterns in Individuals Diagnosed with Conversion Disorder By: Vanessa Frangos Doctorate Candidate University of New Mexico School of Medicine Physical Therapy Program Class of 2013 Advisor: Beth M. Jones, PT, DPT, OCS Printed Name Signature Date Approved by the Program in Physical Therapy, School of Medicine, University of New Mexico in partial fulfillment of the requirements for the degree of Doctor of Physical Therapy. 1 ABSTRACT Background/Purpose: Conversion disorder is a poorly understood diagnosis that is frequently demonized and often misinterpreted as malingering. Conversion disorder is not malingering. Instead it is a coping mechanism of the brain in response to stress. Patients with conversion disorder will present with neurologic-like symptoms, but demonstrate no organic neurological explaination. The purpose of this study is to examine the available literature assessing operant behavioral management of conversion disorder, by physical therapists, in patients presenting with positive motor findings referencing a case study from a personal clinical experience. Case Description: The case report describes a 34-year-old male with an initial presentation of symptoms and diagnosis of Multiple Sclerosis. Mr. E, as he shall henceforth be referenced, was diagnosed with an MS flare, as well as a urinary tract infection (UTI), prior to admission to the hospital for this episode of care. Due to impairments in mobility and self-care, along with medical complexity, which include the UTI, hypertension, and bipolar fisorder, the patient was admitted to inpatient rehabilitation. During his hospitalization, Mr. E was seen by a neurologist and repeat studies were conducted to confirm the MS diagnosis. However, the repeat studies revealed that Mr. E had no spinal cord or brain lesions to suggest a true MS diagnosis, at which point his diagnosis was changed to possible conversion disorder. At the time of discharge the patient’s symptoms continued to wax and wane, as he was prematurely discharged due to current insurance policies. Outcomes: The literature highlights many different treatment options for conversion disorder, which can be grouped as psychiatrically oriented, modality-oriented, or behaviorally oriented1. However, the pertinent evidence is considered to be relatively low ranking and includes: expert opinion, case series, and case reports. Evidence suggests that treatment of conversion disorder is best suited for physical rehabilitation, in conjunction with psychological treatment, in an inpatient setting. The environment can be effectively controlled for relational and circumstantial stressors, as well as facilitate a socially acceptable treatment setting by allied health professionals. Key elements of treatment highlighted are positive and negative behavioral modification techniques, both of which have proven to have astounding functional outcomes. However, in situations where behavioral modification techniques are either not practical or effective at resolving the patients’ symptoms, a variety of other treatments are available. Discussion/Conclusion: Despite the low levels of evidence, the relevant research does demonstrate that physical therapy using behavioral modification techniques can provide effective short-term symptom resolution in patients with positive motor findings, when combined with psychological treatment in an inpatient setting, in a fairly short period of time. 2 TABLE OF CONTENTS ABSTRACT ................................................................................................................................................... 2 CHAPTER 1: BACKGROUND AND PURPOSE ........................................................................................... 4 CHAPTER 2: CASE STUDY ......................................................................................................................... 6 CHAPTER 3: EVIDENCE BASED ANALYSIS ........................................................................................... 11 LITERATURE REVIEW .................................................................................................................. 13 CHAPTER 4: DISCUSSION / CONCLUSION ............................................................................................. 32 REFERENCES ............................................................................................................................................ 34 APPENDICES ............................................................................................................................................. 35 3 CHAPTER 1: BACKGROUND AND PURPOSE At least one-third of physical symptoms that present for medical care are medically unexplained 2. Conversion disorder is one such condition and has many definitions, but in general it is the presence of one or more neurological-like symptoms affecting voluntary motor or sensory function3. The diagnosis was historically termed ―hysteria‖ as its cause was believed to be only in women and due to a wondering uterus. However, it was later determined to affect males and females equally and was consequently renamed. Hysteria became ―conversion disorder‖ based upon the concept that it was triggered by an unconscious psychological mechanism, which ―converts‖ conflicting aspects of an individual’s mental life into neurological signs and symptoms 3. Conversion disorder is currently classified under the Diagnostic and Statistical Manual for psychiatry as a subtype of Somatoform Disorder 4. Conversion disorder is present in between 11- 48 per 100,000 people in the general population 3. Typical symptom presentation includes either positive motor findings, like tremor, dystonia, and gait disturbance; or negative motor findings, like paralysis, or diminished normal movement patterns. Presentation of these symptoms suggests a neurological condition, however, consistent neurological or musculoskeletal pathologies or patterns are absent 3. While the cause of conversion disorder is unknown and the underlying brain mechanisms remain uncertain, current literature suggests that the main focus of treatment should not be on why the symptoms are occurring but rather on how to resolve them. An initial treatment hurdle involves overcoming the patients’ anger about being given a psychiatric diagnosis when they consider their problem to be entirely physical. It is important to remember that conversion symptoms are not under the patient’s voluntary control and instead, are due to an unconscious expression of a psychological conflict or need 3. Potential contributors to motor conversion disorder may include both positive and negative emotional events, major personally relevant crises or minor repeated daily stressors 5. Based on these 4 contributors, it is easily understood why the primary recommendation for treatment is psychologically founded. However, in recent years numerous reports have suggested the need for physical therapy management of this disorder, specifically suggesting that psychological treatment alone is not effective 3. Disability, as a result of conversion disorder, is similar to disability resulting from organic causes in that it affects occupation and social aspects of the person’s life 6. As a consequence, the person often requires comprehensive assessment, treatment and rehabilitation. When the physical symptoms are not addressed, secondary complications like muscle weakness and joint contracture occur in addition to the already disabling physical symptoms. Appropriate functional management is vital for alleviating the conversion symptoms and preventing complications from physical dysfunction 6. While use of physical therapy as treatment for conversion disorder is apparent, it is not funded by most insurances. Furthermore, it remains unclear the exact approach physical therapist should take to yield the best outcomes for their patients. A case study of a 34 year-old male with diagnosed conversion disorder, presenting with positive motor findings, treated by a multidisciplinary team, including physical therapy, is reported in conjunction with this literature analysis is to answer the following clinical question: In adults diagnosed with conversion disorder, presenting with positive motor symptoms, will operant behavioral management of movement patterns, by physical therapists, provide effective symptom resolution better than psychiatric management alone? 5 CHAPTER 2: CASE STUDY Introduction The case subject is a 34-year-old male, married, who lives at home with his wife of 13 years, and their 8-year-old son. Mr. E, as he shall henceforth be referred, was seen for his most recent episode of care, in the Emergency Department in October 2012, after a fall in which he hit his back on the step of his wheelchair and his head on the floor. He developed new onset back and neck pain that required medical attention. Mr. E reported that he was experiencing increased extremity weakness and general decline two days prior to the fall. Mr. E was diagnosed with a Multiple Sclerosis (MS) flare up, two days prior to admission by his primary care physician, and subsequently diagnosed and treated for a urinary tract infection (UTI). Due to impairments in mobility and self-care, along with medical complexity including the UTI, hypertension, and bipolar disorder, the patient was admitted to the hospital for inpatient rehabilitation. In rehabilitation, he was treated with a multidisciplinary approach including physiatry, rehabilitation nursing, physical therapy, occupational therapy, recreational therapy, and social work for discharge planning. Examination Mr. E’s past medical history, pertinent to his MS diagnosis, began 3 years ago. In February 2009, Mr. E was evaluated for complains of headache and symptoms of left-sided weakness and numbness. Symptoms were reported to be ―purely functional‖ as a MRI scan of the brain and cervical regions were negative for neurological findings. In June 2009, patient presented again with similar symptoms, noted for ―breakaway weakness‖, and negative MRI findings. Nine months later, March 2010, Mr. E was readmitted and evaluated for a neurogenic bladder that was subsequently treated with a supra-pubic catheter prior to discharge. Spinal tap with a CSF MS panel was completed in October 2010, but results continued to be classified as normal. Symptoms reoccurred, with increased weakness in his lower extremities in November 2011, again with a normal MRI. In March 2012, Mr. E sought treatment from an outside facility. MRI scans were performed with an Open MRI machine, and several lesions were noted in his brain and spinal cord, 6 resulting in an official diagnosis of MS. The open MRI images were requested and reviewed during this episode of care. Nevertheless repeat studies were conducted by the admitting hospital, including a MRI of the cervical and brain regions, and a CSF MS panel, all of which yielded normal results with no lesions, or other indications to explain Mr. E’s symptoms. Various practitioners completed Mr. E’s physical examinations, all noting non-significant findings for cardiovascular, pulmonary, and abdominal systems. However, there are notable inconsistencies between clinicians for the remaining systems to include: musculoskeletal, coordination, sensory, strength, and reflexes. Furthermore, anatomical patterning was also absent or inconsistent between examinations. One clinician reports ―gross sensory loss to the bilateral face, upper arms, and lower extremities‖ bilaterally, severe spasticity with intentional movements, normal resting tone in all extremities, and hyperreflexive responses, 3+, at the biceps tendon, brachioradialis, and patellar regions bilaterally. Meanwhile the neurology examination yielded the following findings; sensory loss at approximately T9, but not necessarily reproducible, more rigid tone than noted in true spasticity, a non cerebellar tremor that improves as he reaches his endpoint, ―significant break away type weakness‖, noting difficulty evaluating ―true weakness‖ in manual muscle testing, and ―brisk but symmetrical‖ reflexes, 2 to 3+. A psychiatry consult was requested to explore alternate diagnoses based on inconsistencies in the medical examinations and lack of physical evidence to indicate a true MS diagnosis. The Psychiatric Evaluation Team saw Mr. E in November 2012, and per their evaluation, Mr E. was considered to have risk factors associated with the onset of conversion disorder. Mr E’s father committed suicide when he was 5 years old, while his mother was pregnant with his younger brother. Her reports taking his little brother under his wing, and feeling a bit envious of the other children in town whose fathers would take them to do things, as he did not have a father to do similar things with. He eventually went on to finish high school, and start college, but shortly there after was diagnosed with Bipolar Disorder, manifested by a manic episode. Mr. E reports he was admitted to a psychiatric facility for two weeks. He 7 currently denies any associated depression, suicidal ideation, or psychosis since. However, he does admit that he is currently quite frustrated and confused because he recently lost his job, and believed that he was on short-term disability but recently discovered that he was not. By observation Mr. E has a complex and unsupportive relationship with his wife, as she was witnessed reprimanding patient about being a ―terrible father‖ and a ―faker‖. Evaluation Mr. E presents with very strange neurological symptoms. He has a tremor, but it is non-cerebellar, as it actually improves as he reaches his endpoint. There is a similar type of tremor in his upper and lower extremities, which is fairly gross and seems to fluctuate in intensity and frequency. However, is no clinical or diagnostic information to suggest that Mr. E has Multiple Sclerosis. On November 1, 2012, Mr. E’s diagnosis was changed from MS to Conversion Disorder (ICD-9-CM 300.11). The new diagnosis of conversion disorder is not specifically listed in the Guide to Physical Therapist Practice. Nevertheless, the symptoms experienced by Mr. E, weakness, balance/strength difficulties, and in-coordination, do fit under the practice pattern 5A: Primary prevention and risk reduction for loss of balance and falling. While it was difficult for Mr. E, and his family to accept the new diagnosis, it also brought with it hope. MS is a progressive disease with a fairly discouraging prognosis, while conversion disorder bears with it the possibility of a full recovery. While a full recovery is possible, it depends on several variables including family support, treatment approach, financial availability to pay for treatment, and intrinsic desire to move away from the ―sick role‖ in life to a healthy one. Mr. E, unfortunately, does not have a strong support system, his insurance did not cover inpatient physical therapy as a means of treatment, nor did it include coverage for mental health. He recently lost his job creating an incredible financial strain, which increased his psychological stress, and possibly even exacerbated his condition. Finally, Mr. E himself is the only one who can truthfully assess his intrinsic desire to recover. Prognosis for full recovery is poor for MR. E. However, improvement in functional independence can be improved upon as the activity 8 restrictions placed on his life by MS are now lifted. Adaptations were made to his physical therapy sessions for the remainder of his stay to encourage normal movement and build confidence and self worth. Interventions The rehabilitation team treated Mr. E under the MS diagnosis for 16 of his 21-day stay with special care given to prevent over fatigue, and over heating, while addressing his weakness, balance/strength difficulties, and in-coordination. Once his diagnosis was changed to conversion disorder, the approach to physical therapy immediately changed to include; encouragement of gait training with body weight support, use of a manual wheelchair over his power wheelchair for mobility, and discontinued use of his orthoses. Positive reinforcement of progress and effort was provided as often as possible to include reinforcement of the idea that a full recovery is now possible. Unfortunately Mr. E’s insurance did not cover inpatient treatment for conversion disorder, and he was discharged from the hospital after only 4 days of treatment, as treatment is not provided on discharge days. Given the complexity of Mr. E’s case, it was recommended that he seek a second opinion from a multispecialty center such as the Mayo Clinic. Outcomes Functionally Mr. E reported upon admission that he was easily fatigued, unable to walk more than 15 steps on most days, and had required a power wheelchair for most mobility for the last year. He wore a left wrist splint to help with contractures of the left hand, and had bilateral AFO’s, and KAFO’s available as needed to accommodate varying levels of symptom exacerbation. From a therapy stand point, at the time of discharge the patient progressed from moderate assistance to supervision with rolling supine to sit and sit to supine, and minimum assist to supervision for a low pivot transfer from bed/mat table to his wheelchair. Dependent to modified independent for toileting/toilet transfers, from moderate assistance to modified independent with bathing and moderate assistance to modified independent with dressing. Complete scores from Functional Independence Measure (FIM) in Table 1. While Mr. E demonstrates improved functional mobility at the time of discharge, his full rehab potential was not achieved. 9 KEY - Scores Reported by Occupational Therapy Speech Therapy Physical Therapy 10 CHAPTER 3: EVIDENCE BASED ANALYSIS Methods An extensive evidence based literature search was conducted for the literature review. The summary of the search can be found in Figure 1. Four different databases, which included PubMed®, CINAHL Plus, Cochrane, and ISI Web of KnowledgeSM, were used in order to search for the answer to the clinical question. A keyword search was conducted in each database with various combinations of the following key words: Conversion Disorder, Somatoform Disorder, Behavioral Management, and Positive Motor Symptoms. Limits were placed on the initial keyword search to include only articles available in full text, composed in English, and conducted with human subjects. The total number of collected articles was 169. Of the 169 articles, 45 were selected based on the appropriateness/relevance of the title in relation to the PICO question. Thirty-seven articles were excluded for one of the following reasons; the topic was not closely related to the PICO question, or the patient population was not similar. The 8 remaining articles were reviewed and analyzed using article analysis worksheets (see Appendix A). Each article was then assigned an Oxford Levels of Evidence and the Physiotherapy Evidence Database (PEDro) score if appropriate, and summarized in the Literature Review section. 11 Figure 1. Articles Included and Excluded for Analysis PubMed CINAHL Cochrane Web of Knowledge Some keywords: CD or SD; CD or SD & Behavioral Management 76 27 3 63 Keyword Search Keyword Search One keyword Keyword Search One keyword combination Title Search Searched cited references 1 10 Two Different keyword combinations & 5 limits 19 combinations & 3 limits 15 Selected based on appropriateness/ relevance of title in relation to PICO question 45 37 Excluded: Total Articles (Relevant to Topic) Case Report 4 + Literature Review 2 + Retrospective Cohort Study 1 12 Topic not closely related, Non-similar population + Experimental Study 1 = 8 Total Studies -Reviewed -Analyzed -Compared to PICO Literature Review 1. Campo, J. V., & Negrini, B. J. (2000). Case study: Negative reinforcement and behavioral management of conversion disorder. Journal of the American Academy of Child and Adolescent Psychiatry, 39(6), 787-790. Level of Evidence: 5 – Case Report Pedro Score: N/A Purpose: Most researchers have emphasized positive reinforcement for healthy behavior, involving minimizing the rewards associated with the ―sick role‖ they have taken on. Less well described has been the use of negative reinforcement. In this case study, completed on a 12-year-old boy, diagnosed with conversion disorder, after 3 months of persistent right arm pain and immobility. He failed conservative treatment of ibuprofen and physical therapy. A psychological consult reported significant psychological stressors in the past year to include the following; his mother was diagnosed with thyroid cancer, his father lost job and was being treated by a physical therapist for arm pain, he recently received D’s in several subjects at school and was receiving special education services for reading. The purpose is not clearly stated, however it can be deduced that the purpose of this paper is to explore negative reinforcement as a component of behavioral modification techniques to treat his conversion disorder. Methods: It was recommended that strict bed rest be maintained when the subject was not performing critical functions, like using the bathroom, eating, or directly involved in physical therapy, until the symptoms resolved. The subject was placed on a no stimulation protocol which did not allow reading or television, with the rationale being that all of his energy needed to be focused on getting well. 13 Results: After several hours of the no stimulation protocol, the subject excitedly informed his parents that he was regaining some mobility in his arm. His parents were reassuring, but they sent him back to his room to continue resting. Within 24 hours, he had regained complete mobility of his arm without any pain Critique/Bottom Line: This case study is a relatively low level of evidence with an n of 1. Relevant background and research is presented with emphasis on positive reinforcement effectiveness, with a lack of pertinent research on negative reinforcement, and effectiveness in children under the age of 18. This case study illustrates how behavioral interventions, incorporating the use of negative reinforcement, can be just as powerful in treating conversion disorder as positive reinforcement. It can also be seamlessly implemented at a low cost with a rehabilitative approach that encourages a return to usual activities and discourages sick-role behaviors. This treatment approach involves significant familial support to implement, and reinforce compliance. Typically those with conversion disorder do not have this much support, as the immediate family is almost always contributors to the initial onset of symptoms. Consequently this approach in the home setting may be limited based on familial support. 14 2. Ness, D. (2007). Physical therapy management for conversion disorder: Case series. Journal of Neurological Physical Therapy, 31, 30-39. Level of Evidence: 4 – Retrospective Case Series Pedro Score: N/A Purpose: Healthcare providers may be unaware of the important role of physical therapy in the management of patients with conversion disorder. While numerous reports have suggested the need for physical therapy management of this disorders, there is a lack of reports outlining specific physical therapy management principles or daily treatment progression. The purpose of this series of case reports is to increase awareness of conversion disorder and to provide interventions by proposing patient management guidelines. Methods: Retrospective chart review of 3 female patients, between the ages of 18-35, all presenting with positive motor symptoms, carrying a diagnosis of conversion disorder, with a history of abuse and a stressful life event that preceded the onset of their symptoms. Treatment consisted of 3-5 hours per day, 6-7 days a week of physical, occupational and speech therapies in an inpatient setting. Behavioral modification techniques were used throughout all therapies, consisting of ignoring abnormal movement patterns and reinforcing correct movement patters using feedback and praise. Functional Independence Measure (FIM) scores were obtained at admission, discharge, and 3-month follow up. Results: All three patients demonstrated rapid improvement in function within their 8-day mean length of stay in inpatient rehabilitation. The mean discharge FIM score of the three patients was 124/126, with a mean gain in score from admission to discharge of 48 points. All three patients gained complete return of physical functioning and were symptom free at the 3-month follow up. 15 Critique/Bottom Line: This retrospective case series does not note how the patients were selected (inclusion/exclusion criteria, how many cases were reviewed etc.) which challenges the internal validity of the study. Furthermore with an n of 3, the external validity and application to similar populations is also limited. However, due to the social acceptance of physical therapy intervention vs. psychological treatment alone, the feasibility and willingness of patients to participate increases the effectiveness of the intervention. It allows the individual to move away from the ―sick role‖ and to return to healthy roles socially, and physically with gentle guidance through re-establishment of normal movement patterns. Despite the small sample size and lack of comparison using just psychological treatment, this intervention is low in cost, can be implemented within the normal treatment parameters for inpatient rehab, and decreases length of stay with outcomes lasting a minimum of 3 months. Table 2: Functional Independence Measure, Ness D. 16 3. Oh, D., Yoo, E., & Yi, C. (2005). Case report: physiotherapy strategies for a patient with conversion disorder presenting abnormal gait. Physiotherapy Research International, 10(3), 164168. Level of Evidence: 5 – Case Report Pedro Score: N/A Purpose: The objectives of this paper are to describe a structured physiotherapy procedure for a patient with gait disturbance secondary to conversion disorder and then to report the result of behavior modification techniques. This article showcases a case report of a 20-year-old male diagnosed with gait disturbances and balance abnormalities attributed to conversion disorder. Preliminary psychology reports suggested that the subject was ―mentally immature, and lacked adaptability to different situations‖. At the time of treatment the subject was 2 months away from recruitment to the Army. Methods: The subject was given a full explanation of the functional training process before the start of treatment, and goals were set. He was not allowed to walk except during functional training. At all other times he was required to use a wheelchair for mobility. Treatment sessions consisted of individual two hour functional training twice a day, 5 days a week, and a single hour session on Saturdays, for 5 weeks. The subject’s abnormal behavior and communication were ignored, and appropriate skills were rewarded with positive verbal responses. He was rewarded with freedom to walk around the hospital ward when he was able to ambulate normally. Results: The Gait Abnormality Rating Scale (GARS) was used to assess gait function at admission and discharge. After 5 weeks of functional rehab and gait training, the subject demonstrated an improvement in the total 17 GARS score from 22, at initial assessment, to 4 at discharge. He continued to improve for two weeks after discharge, but continued follow up was unavailable because he was recruited to military service. Critique/Bottom Line: Case reports are Level 5 evidence, and lack both external and internal validity. However, the systematic structured functional rehabilitation, combined with behavior modification techniques utilized in this case study appear to be helpful in reducing symptoms and restoring normal function in this patient, and could reasonably be applied to others with similar symptoms. The principle of providing motivation and reducing reinforcement of abnormal movement is a cost effective and powerful way to encourage a transition back to a ―healthy‖ role in life. Especially, if the patient is already being treated in the inpatient setting, by a multidisciplinary team. 18 4. Rosebush, P., & Mazurek, M. (2011). Treatment of conversion disorder in the 21st century: have we moved beyond the couch?. Current Treatment Options in Neurology, 13(3), 255-266. Level of Evidence: 3a - Literature review (not a systematic review) Pedro Score: N/A Purpose: There is no Class I evidence supporting the efficacy of any treatment for conversion disorder; most information is at the Class IV level— expert opinion, case series, case reports, and open studies. While the purpose of this study is not clearly stated, one may deduce that the purpose is to evaluate current treatment options for conversion disorder, their effectiveness, and barriers to treatment, to identify key elements that increase the success of treatment. Methods: Relevant information from current literature was cited on psychotherapy, hypnotherapy, narcotherapy or abreaction, and pharmacotherapy. The method for selecting literature reviewed was not specifically stated. Results: Various treatment options are available for treating conversion disorder, however regardless of treatment, the healthcare provider must be supportive, open-minded, and create a nonjudgmental environment that will allow in-depth inquiry and exploration. This alleviates the patients suspicion that their physicians suspect he or she may be consciously feigning symptoms, and allows the patients specific concerns to be addressed. It is also important to ensure that all appropriate medical and neurologic investigations have actually been carried out, maintaining an open mind to the possible need for further tests, especially if new symptoms develop. Critique/Bottom Line: While this literature review did not specifically denote the search methods, it does explicitly name numerous treatment options and relevant research available on the respective treatment option. Regardless of the 19 treatment option utilized the key appears to lie with the attitude of the healthcare provider. Treatment begins with the healthcare provider and their willingness to add conversion disorder as a real possibility to the differential diagnosis in a timely manner. There is mounting evidence to suggest that patients with an earlier diagnosis and treatment have a greater chance of reversal of symptoms regardless of the treatment. This article suggests that goals of the diagnostic assessment should include identification of the psychosocial circumstances in which the conversion symptoms first occurred and then determine which patients are likely to benefit from psychological exploration and insight-oriented psychotherapy as opposed to more physically based interventions and modalities such as physiotherapy, deep breathing and relaxation techniques. There are a variety of treatment options available to treat conversion disorder based on the individual needs of the patient. Selecting the correct treatment approach may effect the outcome and prognosis. 20 5. Speed, J. (1996). Behavioral management of conversion disorder: Retrospective study. Archives of Physical Medicine and Rehabilitation, 77, 147-154. Level of Evidence: 2b – Retrospective case series Pedro Score: N/A Purpose: Physiatrists have seen patients with conversion disorder for years during the initial evaluation stages due to the nature of the disorder and the corresponding gait and functional deficits. However, it is only recently that physiatrists have been allowed to continue treating patients post diagnosis of conversion disorder. Typically when organic causes of the symptoms have been ruled out patients are referred to psychiatry. The purpose of this study is to assess whether operant behavioral treatment of conversion disorder by physical therapists provides effective and durable symptom resolution, and to evaluate the prognostic value of duration of symptoms, as to the time required to effect symptom resolution. Methods: A retrospective case series investigation was conducted on the first 10 patients, seen by the author, for gait abnormalities secondary to diagnosed conversion disorder. All patients were treated with behavioral modification techniques to include confinement to a wheelchair at all times when not directly involved in therapy, no correction or mention of abnormal gait patterns, and consistent copious positive reinforcement from all members of the interdisciplinary therapy team. Subjects were then evaluated monthly for 7 to 36 months as available. One subject was unavailable immediately after discharge, thus follow up reports were not feasible. Furthermore, the study reviewed 18 articles and summarized their findings regarding various treatment approaches for conversion disorder. Results: All 10 subjects made significant gains in FIM gait scores demonstrating effectiveness of behavior treatment of conversion disorder. However, treatment effect was only effective over long periods of time for 7 of the 9 21 patients available for follow up. Critique/Bottom Line: Limitations of this study include its retrospective, non-blinded design, and the fact that different observers collected FIM ambulation scores. Follow-up times were unable to be standardized and sample size and lack of a control group are obvious weaknesses. However, the conclusions drawn from the data appear valid. At least initially, physical therapy intervention using behavior modification techniques can greatly improve functional independence and normalize gait patterns in a relatively short inpatient rehabilitation stay when paired with a multidisciplinary approach. 22 6. Sumathipala , A. (2007). What is the evidence for the efficacy of treatments for somatoform disorders? A critical review of previous intervention studies. Psychosomatic Medicine, 69, 889-900. Level of Evidence: 1A – SR of SR’s and RCT’s Pedro Score: N/A Purpose: Patients presenting with medically unexplained symptoms to include all functional problems, rather than just the sub-groups meeting the operational criteria for somatization disorder, conversion disorder, or symptoms syndromes, were identified for review. This review attempted to answer the question; what is the highest level of evidence available for the efficacy of pharmaceutical and non-pharmaceutical interventions for patients with medically unexplained symptoms and where have these studies been carried out? The aim was to identify the gaps and then to report any recent advances made since the last systematic review. Methods: An extensive literature review was conducted using electronic databases to include: Cochrane library databases (up to 2007), Medline (1966 –2007), PsychINFO (1974 –2006), and EMBASE (1980 –2007). The following key words were used first individually in the abstract, key words, and title: ―medically unexplained symptoms,‖ ―somatisation,‖ ―somatization,‖ then in combination with psychological therapies, cognitive behavior therapy, pharmacological therapies, management, therapy, drug therapy, and antidepressants, and ―somatoform disorders‖. To ensure a comprehensive review, search for literature was supplemented by examining the reference lists of the papers generated from the original searches. Inclusion criteria included Systematic Reviews (SR) and Randomized Controlled Trails (RCT) of psychological, pharmacological, or any other type of intervention involving an adult, with defined medically unexplained symptoms, written in English. Articles were excluded for the following reasons: duplicates, 23 SR’s or RCT’s exclusively on symptom syndromes (irritable bowel syndrome or fibromyalgia), and studies focusing on children or adolescents. Results: Search results from Medline, PsychINFO, and EMBASE did not yield any suitable SR’s. The Cochrane Library yielded 13 relevant SR abstracts, six of which were potentially relevant and were studied in detail. Search results from all 4 databases yielded 108 RCT abstracts, 14 of which were selected for this review. Five systematic reviews have shed light on the use of cognitive behavioral therapy, showing varying success in the management of patients with medically unexplained symptoms, while only one systematic review on antidepressant medication could be effective in improving outcome, including symptoms and disability. Critique/Bottom Line: The review reveals that two types of interventions, antidepressant medication and cognitive behavioral therapy are supported by Level I evidence as benefiting patients with medically unexplained symptoms. There is more Level I evidence for cognitive behavioral therapy compared with other approaches and the evidence is increasing. Cognitive behavioral therapy seems to be effective in the reduction of a wide range of physical symptoms and associated mood disturbance, as well as in producing improvements in overall physical and social functioning. Antidepressants are moderately effective for medically unexplained symptoms and effect sizes are homogenous across functional syndromes but are associated withdrawal symptoms and side effects. There are currently no trials comparing antidepressants with cognitive behavioral therapy. 24 7. Tocchio, S. (2009). Treatment of conversion disorder: A clinical and holistic approach. Journal of Psychosocial Nursing, 47(8), 42-47. Level of Evidence: 5 – Case Report Pedro Score: N/A Purpose: The author of the article reports treating multiple cases of conversion disorder, typically with behavioral modification techniques. However, in this case typical treatment techniques were not displaying the same level of effectiveness demonstrated in prior cases. The purpose of this article is to encourage other healthcare providers to attempt alternative medicine techniques without cynicism, by showcasing the success of one patient with conversion disorder while emphasizing the value of nursing as a part of a team approach. Methods: A 19-year-old woman presented to the inpatient rehabilitation unit with a two-year history of medically unexplained symptoms of spontaneous seizures, paralysis from the mouth down, blindness, inability to ambulate and vomiting after all attempts at eating. The patient had the diagnosis of conversion disorder and was treated with a multidisciplinary approach to include psychiatry, and neurologist. Results: Behavioral modification with set rules and schedules failed to resolve symptoms in this case. Biofeedback, meditation and hypnosis were added to the plan of care. Symptoms resolved, and patient regained ability to ambulate independently prior to discharge. Critique/Bottom Line: Every patient with diagnosed conversion disorder will present with various symptoms and respond to treatments to differently. This particular patient rejected behavioral modification techniques and positive verbal encouragement stating that it ―increased her level of stress‖ and initiated her symptoms more 25 frequently. It is important to consider these variations and keep an open mind to a variety of treatment options to include holistic therapies. This study is limited as it is a single case report with limited external validity. Physical therapy was not specifically mentioned as an element of care in the disciplinary team, however, biofeedback is a tool Physical Therapists can use to assist in treatment of conversion disorder if applicable. 26 8. Voon, V., Brezing, C., Gallea, C., Ameli, R., Roelofs, K., LaFrance Jr, W., & Hallett, M. (2010). Emotional stimuli and motor conversion disorder. Brain, 133, 1526-1536. Level of Evidence: 2b - Individual cohort study (including low quality RCT; <80% f/up) Pedro Score: 4/10 Purpose: Currently there is no way to conclusively diagnose conversion disorder vs. malingering. Literature suggests a potential role between arousal and conversion disorder that may play a role in modulating motor networks, resulting in positive motor symptoms. The purpose of this study is to investigate the relationship between affect, or arousal, and conversion disorder with positive motor symptoms, by investigating amygdala activity in association with viewing affective stimuli, using an affective task that has been extensively investigated in healthy volunteers and in patients with psychiatric disorders. The amygdala was a target of this investigation based on its critical role in modulating motivated attention in preparation for action, and its possible influence on the supplementary motor area downstream, involved in motor initiation and non-conscious response inhibition. Methods: Patients with motor conversion disorder were recruited from the Human Motor Control Section clinic at the National Institute of Neurological Disorders and Stroke, National Institutes of Health (NIH). Inclusion criteria included diagnostic confirmation by at least two neurologists and one psychiatrist, no movement symptoms at rest for the imaging study, movement symptoms not affecting the head or neck, no history of traumatic brain injury, and 19 years of age or older. Exclusion criteria included having a serious medical or neurological illness, current major depression, panic disorder, post-traumatic stress disorder, substance abuse or other major affective or psychotic disorders, being on antidepressants and contraindications for MRI. Functional Magnetic Resonance Imaging (fMRI) study using a block design incidental affective task with fearful, happy, and neutral face stimuli and compared valence contrasts between 16 patients with 27 conversion disorder and 16 age- and gender-matched healthy volunteers recruited from the NIH healthy volunteer database. Results: Behaviorally, there was no difference in reaction time between patients with motor conversion disorder and healthy volunteers and there was no difference in amygdala activity when the neutral condition was compared between patients with motor conversion disorder and healthy volunteers. However, imaging did reveal that patients with motor conversion disorder had greater right amygdala activity compared to healthy volunteers when exposed to happy and fearful stimuli. There was also a significant positive correlation in the path from the right amygdala to the right supplementary motor area during both the happy and the fearful conditions. Thus it can be concluded that motor conversion disorder is characterized by greater amygdala activity to arousal and potential impairments in habituation to arousing stimuli. Critique/Bottom Line: While this article does not specifically address physical therapy intervention to treat conversion disorder, it does provide a way to more conclusively diagnosis conversion disorder. More conclusive diagnosis will allow for better, higher level evidence to be conducted on patients with MRI studies to confirm their diagnosis, as well as encourage further research towards biological or psychological treatments targeting arousal as an alternative form of treatment for conversion disorder. 28 Table 2. Results / Article Summaries # 1 Study & Origin Campo, J.V. and Negrini, B.J. (2000) Oxford Level of Evidence Pedro Score 5 N/A The purpose is not clearly stated, however it can be deduced that the purpose of this paper is to explore negative reinforcement as a component of behavioral treatment of conversion disorder. Amount of upper extremity movement and pain as reported/demonstr ated by case subject. Full resolution of symptoms with in 24 hours. 4 N/A The purpose of this series of case reports is to increase awareness of conversion disorder and to provide interventions by proposing patient management guidelines. Functional Independence Measure (FIM) taken at - Admission - Discharge - 3 Month Follow-up All three patients gained complete return of physical functioning and were symptom free at three-month follow-up. To describe a structured physiotherapy procedure for a patient with gait disturbance secondary to conversion disorder and report the result of treatment. Gait Abnormality Rating Scale (GARS) Significant functional gains were achieved with a improvement in total GARS score, with significant gains in general and lower extremity function. Gains continued to be made after the conclusion of therapy for two weeks after. Continued follow up was unavailable as subject was recruited to military service. US Ness, D. (2007) 2 US Oh, D.W., et. al. (2005) 3 Korea 5 N/A Purpose of Study Outcome Measures 29 Results Accept Results to Answer Clinical Question Yes Yes Statistical significance cannot be determined based on study design, but clinical significance is possible due to low costs to implement and good functional gains in a short period of time. Yes 4 Rosebush, P.I., and Maxurek, M.F. (2011) 3a N/A The purpose of this study is not clearly stated. One may deduce that the purpose is to evaluate current treatment options for CD, their effectiveness, barriers to treatment, and identify key elements that increase success of treatment. Current literature, and its evaluation of specific treatment options It is important to create a supportive, openminded, and nonjudgmental environment that will allow in-depth inquiry and exploration, in which the patients concerns that doctors suspect he or she may be consciously feigning symptoms can be addressed. It is also important to ensure that all appropriate medical and neurologic investigations have actually been carried out, maintaining an open mind to the possible need for further tests, especially if new symptoms develop Yes Speed, J. (1996) 2b N/A To assess whether operant behavioral treatment of conversion disorder provides effective and durable symptom resolution, and to evaluate the prognostic value of duration of symptoms, as to the time required to effect symptom resolution. Functional Independence Measure (FIM) Ambulation Score At least initially, physical therapy intervention using behavior modification techniques, can greatly improve functional independence and normalize gait patterns in a relatively short inpatient rehabilitation stay when paired with a multidisciplinary approach. Yes Sumathipala, A. (2007) 1A N/A This review attempted to answer the question, what is the highest level of evidence available for the efficacy of pharmaceutical and nonpharmaceutical interventions for patients with MUS and where have these studies been carried out? The aim was to identify the gaps and then to report any recent advances made since the last systematic review. Primary outcomes were physical symptoms, psychological distress studies, or functional status but some had more than one primary outcome There is more level I evidence for CBT compared with other approaches and the evidence is increasing. CBT seems to be effective in the reduction of a wide range of physical symptoms and associated mood disturbance, as well as in producing improvements in overall physical and social functioning. Yes Canada 5 6 London 30 Tocchino, S.L. 7 5 N/A The stated purpose of this article is to encourage others by describing the success that psychiatry and alternative medicine can offer to patients with conversion disorder and to emphasize the value of nursing as a part of that team. Symptom resolution by observation, and use of a device for ambulation. 2b 4/10 To investigate the relationship between affect or arousal and conversion disorder with positive motor symptoms by investigating amygdala activity in association with viewing affective stimuli. Functional MRI during neutral, fearful and happy conditions /stimulus. (2009) Voon, V. et. al. (2010) 8 US Reaction time to affective conditions Beck Depression Inventory (BDI) and Beck Anxiety Inverntory (BAI) scores 31 Every patient with diagnosed Conversion Disorder will present with various symptoms and respond to treatments to differently. This particular patient rejected behavioral modification techniques and positive verbal encouragement stating that it increased her level of stress and initiated her symptoms more frequently. It is important to consider these variations and keep an open mind to a variety of treatment options to include holistic therapies. Patients with motor conversion disorder had greater right amygdala activity compared to healthy volunteers when exposed to happy and fearful stimuli, with no difference in amygdala activity when the neutral condition was compared between groups. No No CHAPTER 4: DISCUSSION The majority of the research conducted on conversion disorder, or similar medically unexplained symptoms, are low quality articles with the majority of literature composed based on case reports. However, the research is effective in describing the types of treatments available and currently being utilized in various fields of practice. Treatment options include psychiatry, pharmacologically (antidepressants, antianxiety), biofeedback, and behavioral modification therapy by a variety of practitioners. Each treatment has proven to be effective to some extent, at least initially. However, follow up studies are also limited. Conversion disorder is a complex and variable diagnosis that is difficult to diagnosis conclusively, arduous for some practitioners to assign and treat without bias, and even more laborious to conduct high quality research on. This is an unfortunate truth as many insurance companies assign higher value to better quality studies with long term follow up to fully back a treatment practice. In the case of Mr. E, he was treated for 23 days in the inpatient setting under the false diagnosis and limited treatment intensity of MS. When he was re-diagnosed it was only a matter of days before his insurance benefits were removed and he was prematurely discharged prior to reaching his full rehab potential due to inadequate funds. According to Ness, Mr. E, pending an initial diagnosis of conversion disorder and the use of behavioral modification techniques, could have made a full functional recovery of at least Modified Independent on his FIM’s in just 9 days3. CHAPTER 5: CONCLUSION/ BOTTOM LINE Despite psychiatric care named as the current primary intervention for conversion disorder, the patient’s physical symptoms are still just as real and relevant as they were prior to the official diagnosis. Rosebush suggests that the foremost barrier to treatment is the failure of physicians to include conversion disorder into differential diagnosis in a timely manner4. The delay of the initial diagnosis can affect the prognosis of the patient as they become more and more convinced that the symptoms are in fact physical, 32 and that their medical team has simply given up. One significant delay in diagnosis is due to the inability to conclusively distinguish conversion disorder from malingering. The message patients often hear from their medical team is, ―I don’t really believe your signs and symptoms and, because there is nothing wrong, I’m sending you to a psychiatrist‖4. However because treatment strategies for conversion disorder can also be applied to patients suspected of malingering, with similar outcomes, it can be approached with the same behavioral modification techniques, and presented in a way that is less threatening to the patient. The presentation of the new diagnosis is absolutely critical to facilitating a successful recovery. It is important to reassure the patient that their symptoms are real, and are not under voluntary control, that this is the best diagnosis for their symptoms because it also comes with the chance to recovery fully, and that the medical team is still actively seeking the best path to assist in recovery. A multidisciplinary approach, in an inpatient setting, to include physical therapy, is not just a more socially accepted intervention; it is also justifiable based on evidence for effective symptom resolution. Because patients with conversion disorder place a heavy burden on the health system due to their disproportionate consumption of health resources, effective symptom resolution is critical2. Most insurance companies do not cover an inpatient stay for treatment of conversion disorder, as demonstrated in Mr. E’s case. Nevertheless, physical therapy combined with behavioral modification techniques, using both positive and negative reinforcement, is a cost effective, socially accepted, and low risk form of treatment for conversion disorder. 33 REFERENCES 1. Speed, J. (1996). Behavioral management of conversion disorder: Retrospective study. Archives of Physical Medicine and Rehabilitation, 77, 147-154. 2. Sumathipala , A. (2007). What is the evidence for the efficacy of treatments for somatoform disorders? A critical review of previous intervention studies. Psychosomatic Medicine, 69, 889-900. 3. Ness, D. (2007). Physical therapy management for conversion disorder: Case series. Journal of Neurological Physical Therapy, 31, 30-39. 4. Rosebush, P., & Mazurek, M. (2011). Treatment of conversion disorder in the 21st century: have we moved beyond the couch?. Current Treatment Options in Neurology, 13(3), 255-266 5. Voon, V., Brezing, C., Gallea, C., Ameli, R., Roelofs, K., LaFrance Jr, W., & Hallett, M. (2010). Emotional stimuli and motor conversion disorder. Brain, 133, 1526-1536. 6. Oh, D., Yoo, E., & Yi, C. (2005). Case report: physiotherapy strategies for a patient with conversion disorder presenting abnormal gait. Physiotherapy Research International, 10(3), 164-168. 7. Campo, J. V., & Negrini, B. J. (2000). Case study: Negative reinforcement and behavioral management of conversion disorder. Journal of the American Academy of Child and Adolescent Psychiatry, 39(6), 787-790. 8. Tocchio, S. (2009). Treatment of conversion disorder: A clinical and holistic approach. Journal of Psychosocial Nursing, 47(8), 42-47. 34 APPENDICES Appendix A. Evidence Based Article Appraisal Worksheets Appendix A.1 Intervention – Evidence Appraisal Worksheet Citation: Campo, J. V., & Negrini, B. J. (2000). Case study: Negative reinforcement and behavioral management of conversion disorder. Journal of the American Academy of Child and Adolescent Psychiatry, 39(6), 787-790. Level of Evidence (Oxford scale): 5 – Case Report Is the purpose and background information sufficient? Appraisal Criterion Reader’s Comments Study Purpose The purpose is not clearly stated, however it can be deduced that the purpose of this paper is to explore negative reinforcement, as a component of behavioral treatment of conversion disorder. Stated clearly? Usually stated briefly in abstract and in greater detail in introduction. May be phrased as a question or hypothesis. A clear statement helps you determine if topic is important, relevant and of interest to you. Consider how the study can be applied to PT and/or your own situation. What is the purpose of this study? Literature Most researchers have emphasized positive reinforcement for healthy behavior, involving minimizing the rewards associated with the sick role. Less well described has been the use of negative reinforcement. Negative reinforcement produces an increase in the frequency of a desired response by removing an aversive event immediately after the desired response has been performed Relevant background presented? A review of the literature should provide background for the study by synthesizing relevant information such as previous research and gaps in current knowledge, along with the clinical importance of the topic. Describe the justification of the need for this study 35 Does the research design have strong internal validity? Appraisal Criterion Reader’s Comments N/A. Single subject case study. Discuss possible threats to internal validity in the research design. Include: Assignment Attrition History Instrumentation Maturation Testing Compensatory Equalization of treatments Compensatory rivalry Are the results of this therapeutic trial valid? Appraisal Criterion Reader’s Comments 1. Did the investigators randomly assign subjects to treatment groups? a. If no, describe what was done b. What are the potential consequences of this assignment process for the study’s results? 2. Did the investigators know who was being assigned to which group prior to the allocation? a. If they were not blind, what are the potential consequences of this knowledge for the study’s results? 3. Were the groups similar at the start of the trial? Did they report the demographics of the study groups? a. If they were not similar – what differences existed? b. Do you consider these differences a threat to the research validity? How might the differences between groups affect the results of the study? N/A. Single subject case study. N/A. Single subject case study. N/A. Single subject case study. 36 4. Did the subjects know to which treatment group they were assign? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results 5. Did the investigators know to which treatment group subjects were assigned? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results 6. Were the groups managed equally, apart from the actual experimental treatment? a. If not, what are the potential consequences of this knowledge for the study’s results? 7. Was the subject follow-up time sufficiently long to answer the question(s) posed by the research? a. If not, what are the potential consequences of this knowledge for the study’s results? 8. Did all the subjects originally enrolled complete the study? a. If not how many subjects were lost? b. What, if anything, did the authors do about this attrition? c. What are the implications of the attrition and the way it was handled with respect to the study’s findings? 9. Were all patients analyzed in the groups to which they were randomized (i.e. was there an intention to treat analysis)? a. If not, what did the authors do with the data from these subjects? b. If the data were excluded, what are the potential consequences for this study’s results? Yes, single subject case study. Yes, single subject case study. N/A. Single subject case study. No follow up was completed. The only subject involved completed the study. N/A. Single subject case study. 37 Are the valid results of this RCT important? Appraisal Criterion Reader’s Comments 10. What were the statistical findings of this There is no statistical analysis completed for this study? case study. a. When appropriate use the calculation forms below to determine these values b. Include: tests of differences With p-values and CI c. Include effect size with p-values and CI d. Include ARR/ABI and RRR/RBI with p-values and CI e. Include NNT and CI f. Other stats should be included here 11. What is the meaning of these statistical N/A findings for your patient/client’s case? What does this mean to your practice? 12. Do these findings exceed a minimally N/A important difference? Was this brought up or discussed? a. If the MCID was not met, will you still use this evidence? Can you apply this valid, important evidence about an intervention in caring for your patient/client? What is the external validity? Appraisal Criterion Reader’s Comments 13. Does this intervention sound appropriate for use (available, affordable) in your clinical setting? Do you have the facilities, skill set, time, 3rd party coverage to provide this treatment? This treatment approach involves significant familial support to implement, and typically those with CD do not have this much support, as the immediate family is almost always contributors to the initial onset of symptoms. Consequently this approach may be limited based on familial support. However it is cost effective, and not time intensive from the physical therapy stand point. 14. Are the study subjects similar to your patient/ client? a. If not, how different? Can you use this intervention in spite of the differences? I do not typically work with pediatric cases, but based on the conversion disorder diagnosis and the common presence of CD in adolescence it is not unlikely to see similar subjects in my setting. 38 15. Do the potential benefits outweigh the potential risks using this intervention with your patient/client? The subject was still able to complete his basic needs of eating and toileting, as well as get supplementary mobility exercises and treatment with physical therapy preventing unnecessary atrophy and decreased ROM of uninvolved limbs. In the event that the intervention was not yielding powerful results within a week or so it would be necessary to try a different approach to avoid unnecessary deconditioning. 16. Does the intervention fit within your patient/client’s stated values or expectations? a. If not, what will you do now? Yes. The subject presented for treatment with expectations to get better. 17. Are there any threats to external validity in this study? The main threat to external vailidity is the low sample size which prevents the results from being applicable to a larger population. What is the bottom line? Appraisal Criterion Reader’s Comments PEDRO score N/A Summarize your findings and relate this back to clinical significance Behavioral interventions incorporating the use of negative reinforcement can be just as powerful in treating conversion disorder, as positive reinforcement. It can also be easily implemented at a low cost in addition to a rehabilitative approach that encourages a return to usual activities and discourages sick-role behaviors. 39 Appendix A.2 Intervention – Evidence Appraisal Worksheet Citation: Ness, D. (2007). Physical therapy management for conversion disorder: Case series. Journal of Neurological Physical Therapy, 31, 30-39. Level of Evidence (Oxford scale): 4 – Retrospective Case Series Is the purpose and background information sufficient? Appraisal Criterion Reader’s Comments Study Purpose The purpose of this series of case reports is to increase awareness of conversion disorder and to provide interventions by proposing patient management guidelines. Stated clearly? Usually stated briefly in abstract and in greater detail in introduction. May be phrased as a question or hypothesis. A clear statement helps you determine if topic is important, relevant and of interest to you. Consider how the study can be applied to PT and/or your own situation. What is the purpose of this study? Literature The incidence of individuals with conversion disorder has been reported to be between 11 and 48 per 100,000 people in the general population. Conversion disorder can occur in individuals of any age, race, ethnic, or social background. Some studies report a higher frequency in women than in men other studies have found no difference between the sexes. There tends to be a higher incidence of first-degree relatives with psychiatric or medical disorders in individuals with conversion disorder. An association also has been found with conversion disorder and a history of sexual or physical abuse. Recent work suggests psychological treatment alone is not effective in treating conversion disorder Relevant background presented? A review of the literature should provide background for the study by synthesizing relevant information such as previous research and gaps in current knowledge, along with the clinical importance of the topic. Describe the justification of the need for this study 40 Does the research design have strong internal validity? Appraisal Criterion Reader’s Comments Discuss possible threats to internal validity in the research design. Include: Assignment Attrition History Instrumentation Maturation Testing Compensatory Equalization of treatments Compensatory rivalry Statistical Regression Study design: retrospective chart review. The article does not note how the patients were selected (inclusion/exclusion criteria, how many cases were reviewed etc. ) and this significantly challenges the internal validity of the study. Are the results of this therapeutic trial valid? Appraisal Criterion Reader’s Comments 1. Did the investigators randomly assign subjects to treatment groups? a. If no, describe what was done b. What are the potential consequences of this assignment process for the study’s results? 2. Did the investigators know who was being assigned to which group prior to the allocation? a. If they were not blind, what are the potential consequences of this knowledge for the study’s results? 3. Were the groups similar at the start of the trial? Did they report the demographics of the study groups? a. If they were not similar – what No. Retrospective case study, random assignment into groups does not apply. Researchers were not blinded as to the selection of cases to be reviewed. Cases selected were similar with positive motor findings and diagnoses of CD, and all were treated similarly. 41 4. 5. 6. 7. differences existed? b. Do you consider these differences a threat to the research validity? How might the differences between groups affect the results of the study? Did the subjects know to which treatment group they were assign? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results Did the investigators know to which treatment group subjects were assigned? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results Were the groups managed equally, apart from the actual experimental treatment? a. If not, what are the potential consequences of this knowledge for the study’s results? Was the subject follow-up time sufficiently long to answer the question(s) posed by the research? a. If not, what are the potential consequences of this knowledge for the study’s results? No control studies reviewed. No. Retrospective case study, blinding subjects does not apply. Investigators were not blinded to treatment applied when assessing outcomes. No control cases included. Subject follow up time was 3 months, and is sufficient to answer short-term efficacy of treatment. Future studies should evaluate treatment technique and long term efficacy of treatment to determine benefits to decreasing cost in health care 8. Did all the subjects originally enrolled Unable to determine based on information given. complete the study? a. If not how many subjects were lost? b. What, if anything, did the authors do about this attrition? c. What are the implications of the attrition and the way it was handled with respect to the study’s findings? 9. Were all patients analyzed in the groups No control cases included. to which they were randomized (i.e. was there an intention to treat analysis)? a. If not, what did the authors do with the data from these subjects? 42 b. If the data were excluded, what are the potential consequences for this study’s results? Are the valid results of this RCT important? Appraisal Criterion Reader’s Comments 10. What were the statistical findings of this study? a. When appropriate use the calculation forms below to determine these values b. Include: tests of differences With p-values and CI c. Include effect size with p-values and CI d. Include ARR/ABI and RRR/RBI with p-values and CI e. Include NNT and CI f. Other stats should be included here No statistical data presented. 11. What is the meaning of these statistical N/A findings for your patient/client’s case? What does this mean to your practice? 12. Do these findings exceed a minimally N/A important difference? Was this brought up or discussed? a. If the MCID was not met, will you still use this evidence? Can you apply this valid, important evidence about an intervention in caring for your patient/client? What is the external validity? Appraisal Criterion Reader’s Comments 13. Does this intervention sound appropriate for use (available, affordable) in your clinical setting? Do you have the facilities, skill set, time, 3rd party coverage to provide this treatment? 14. Are the study subjects similar to your patient/ client? a. If not, how different? Can you use this intervention in spite of the differences? Yes. While it is not an exceptional study the intervention is low cost, time effective, and correlates well with current PT interventions. Yes. In the neurological setting it is not uncommon to run across medically unexplained conditions or specifically classified conversion disorder. 43 15. Do the potential benefits outweigh the potential risks using this intervention with your patient/client? Intervention has no foreseeable associated risks. In fact, it would be worse to withhold physical therapy intervention in the event of misdiagnosis, in which a person without CD and a undiagnosed neurologic condition, than to treat CD and continue to rule out a true neurological condition. 16. Does the intervention fit within your patient/client’s stated values or expectations? a. If not, what will you do now? Yes. Patients with true conversion disorder typically do want to get better. 17. Are there any threats to external validity in this study? Yes. Due to the nature of conversion disorder, the difficulty diagnosing it, and the inability to truly distinguish it from malingering, as well as, the poor design specification and sample size. What is the bottom line? Appraisal Criterion Reader’s Comments PEDRO score N/A Summarize your findings and relate this back to clinical significance Physical therapy is a more socially accepted intervention or cure to their illness, than psychological treatment alone. It allows the individual to move away from the ―sick role‖ and to return to healthy roles socially, and physically with gentle guidance through re-establishment of normal movement patterns. Despite the small sample size and lack of comparison using just psychological treatment, this intervention is low in cost, can be implemented within the normal treatment parameters for inpatient rehab, and decreases length of stay with lasting effects. 44 Appendix A.3 Intervention – Evidence Appraisal Worksheet Citation: Oh, D., Yoo, E., & Yi, C. (2005). Case report: physiotherapy strategies for a patient with conversion disorder presenting abnormal gait. Physiotherapy Research International, 10(3), 164-168. Level of Evidence (Oxford scale): 5 – Case Report Is the purpose and background information sufficient? Appraisal Criterion Reader’s Comments Study Purpose The objectives of this paper are to describe a structured physiotherapy procedure for a patient with gait disturbance secondary to conversion disorder and then to report the result of treatment. Stated clearly? Usually stated briefly in abstract and in greater detail in introduction. May be phrased as a question or hypothesis. A clear statement helps you determine if topic is important, relevant and of interest to you. Consider how the study can be applied to PT and/or your own situation. What is the purpose of this study? Literature Relevant background presented? A review of the literature should provide background for the study by synthesizing relevant information such as previous research and gaps in current knowledge, along with the clinical importance of the topic. Describe the justification of the need for this study Yes, especially relevant to the application of physical therapy as a primary treatment to address physical symptoms of CD. The study states that the treatment of conversion disorder is well suited to the physical rehabilitation setting because of the potential for successful environmental control, and because of the experience of the multidisciplinary team in treating analogous organic conditions with similar types of functional losses 45 Does the research design have strong internal validity? Appraisal Criterion Reader’s Comments N/A. Single subject case study. Discuss possible threats to internal validity in the research design. Include: Assignment Attrition History Instrumentation Maturation Testing Compensatory Equalization of treatments Compensatory rivalry Statistical Regression Are the results of this therapeutic trial valid? Appraisal Criterion Reader’s Comments 1. Did the investigators randomly assign subjects to treatment groups? a. If no, describe what was done b. What are the potential consequences of this assignment process for the study’s results? 2. Did the investigators know who was being assigned to which group prior to the allocation? a. If they were not blind, what are the potential consequences of this knowledge for the study’s results? 3. Were the groups similar at the start of the trial? Did they report the demographics of the study groups? a. If they were not similar – what differences existed? b. Do you consider these differences a threat to the N/A. Single subject case study. N/A. Single subject case study. N/A. Single subject case study. 46 4. 5. 6. 7. 8. 9. research validity? How might the differences between groups affect the results of the study? Did the subjects know to which treatment group they were assign? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results Did the investigators know to which treatment group subjects were assigned ? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results Were the groups managed equally, apart from the actual experimental treatment? a. If not, what are the potential consequences of this knowledge for the study’s results? Was the subject follow-up time sufficiently long to answer the question(s) posed by the research? a. If not, what are the potential consequences of this knowledge for the study’s results? Did all the subjects originally enrolled complete the study? a. If not how many subjects were lost? b. What, if anything, did the authors do about this attrition? c. What are the implications of the attrition and the way it was handled with respect to the study’s findings? Were all patients analyzed in the groups to which they were randomized (i.e. was there an intention to treat analysis)? a. If not, what did the authors do with the data from these subjects? b. If the data were excluded, what are the potential consequences for this study’s results? Yes, single subject case study. Yes, single subject case study. N/A. Single subject case study. Follow up was limited to two weeks due to recruitment to the military. The only subject involved completed the study. N/A. Single subject case study. 47 Are the valid results of this RCT important? Appraisal Criterion Reader’s Comments 10. What were the statistical findings of this There is no statistical analysis completed for this study? case study. a. When appropriate use the calculation forms below to determine these values b. Include: tests of differences With p-values and CI c. Include effect size with p-values and CI d. Include ARR/ABI and RRR/RBI with p-values and CI e. Include NNT and CI f. Other stats should be included here 11. What is the meaning of these statistical N/A findings for your patient/client’s case? What does this mean to your practice? 12. Do these findings exceed a minimally N/A important difference? Was this brought up or discussed? a. If the MCID was not met, will you still use this evidence? Can you apply this valid, important evidence about an intervention in caring for your patient/client? What is the external validity? Appraisal Criterion Reader’s Comments 13. Does this intervention sound appropriate for use (available, affordable) in your clinical setting? Do you have the facilities, skill set, time, 3rd party coverage to provide this treatment? Yes, I believe that inpatient rehab is the best place to treat CD due to environmental control, and because of the experience of the multidisciplinary treatment in treating analogous organic conditions with similar types of functional losses. Yes. In the neurological setting it is not uncommon to run across medically unexplained conditions or specifically classified conversion disorder. 14. Are the study subjects similar to your patient/ client? a. If not, how different? Can you use this intervention in spite of the differences? 15. Do the potential benefits outweigh the potential risks using this intervention with your patient/client? The potential benefit of treatment is full recovery where the risk of not using this intervention would be a possible increase in muscle weakness and joint contracture. 48 16. Does the intervention fit within your patient/client’s stated values or expectations? a. If not, what will you do now? Yes. Patients with true conversion disorder typically do want to get better. 17. Are there any threats to external validity in this study? Yes. Due to the nature of conversion disorder, the difficulty diagnosing it, and the inability to truly distinguish it from malingering, it is difficult to ascertain that the treatment is the determining factor in recovery. What is the bottom line? Appraisal Criterion Reader’s Comments PEDRO score N/A Summarize your findings and relate this back to clinical significance The systematic structured functional rehabilitation, combined with behavior modification techniques utilized in this case study appear to be helpful in reducing symptoms and restoring normal function in this patient, and could reasonably be applied to others with similar symptoms. The principle of providing motivation and reducing reinforcement of abnormal movement, which contributes to the disabled state, is a cost effective and powerful way to encourage return to the ―healthy‖ role in life. 49 Appendix A.4 Intervention – Evidence Appraisal Worksheet Citation: Rosebush, P., & Mazurek, M. (2011). Treatment of conversion disorder in the 21st century: have we moved beyond the couch? Current Treatment Options in Neurology, 13(3), 255-266. Level of Evidence (Oxford scale): 3a - Literature review (not a systematic review) Is the purpose and background information sufficient? Appraisal Criterion Reader’s Comments Study Purpose Stated clearly? Usually stated briefly in abstract and in greater detail in introduction. May be phrased as a question or hypothesis. The purpose of this study is not clearly stated. One may deduce that the purpose is to evaluate current treatment options for CD, their effectiveness, barriers to treatment, and identify key elements that increase success of treatment. A clear statement helps you determine if topic is important, relevant and of interest to you. Consider how the study can be applied to PT and/or your own situation. What is the purpose of this study? Literature There is no Class I evidence supporting the efficacy of any treatment for CD; most information is at the Class IV level— expert opinion, case series, case reports, and open studies. CD patients present with a wide range of neurologic signs and symptoms and are typically referred to psychiatry after investigations fail to yield a medical or neurologic diagnosis that can adequately explain their disability. The cause of CD is unknown and the underlying brain mechanisms remain uncertain. Relevant background presented? A review of the literature should provide background for the study by synthesizing relevant information such as previous research and gaps in current knowledge, along with the clinical importance of the topic. Describe the justification of the need for this study 50 Does the research design have strong internal validity? Appraisal Criterion Reader’s Comments Discuss possible threats to internal validity in the research design. Include: Assignment Attrition History Instrumentation Maturation Testing Compensatory Equalization of treatments Compensatory rivalry Statistical Regression No. Relevant information from current literature was cited on psychotherapy, hypnotherapy, narcotherapy or abreaction, and pharmacotherapy. However, the method for selecting literature reviewed was not specifically stated. At the end of the article it did yield the following disclosure statement: No potential conflicts of interest relevant to this article were reported. Are the results of this therapeutic trial valid? N/A Appraisal Criterion Reader’s Comments 1. Did the investigators randomly assign subjects to treatment groups? a. If no, describe what was done b. What are the potential consequences of this assignment process for the study’s results? 2. Did the investigators know who was being assigned to which group prior to the allocation? a. If they were not blind, what are the potential consequences of this knowledge for the study’s results? 3. Were the groups similar at the start of the trial? Did they report the demographics of the study groups? a. If they were not similar – what N/A N/A N/A 51 4. 5. 6. 7. 8. 9. differences existed? b. Do you consider these differences a threat to the research validity? How might the differences between groups affect the results of the study? Did the subjects know to which treatment group they were assign? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results Did the investigators know to which treatment group subjects were assigned ? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results Were the groups managed equally, apart from the actual experimental treatment? a. If not, what are the potential consequences of this knowledge for the study’s results? Was the subject follow-up time sufficiently long to answer the question(s) posed by the research? a. If not, what are the potential consequences of this knowledge for the study’s results? Did all the subjects originally enrolled complete the study? a. If not how many subjects were lost? b. What, if anything, did the authors do about this attrition? c. What are the implications of the attrition and the way it was handled with respect to the study’s findings? Were all patients analyzed in the groups to which they were randomized (i.e. was there an intention to treat analysis)? a. If not, what did the authors do with the data from these subjects? N/A N/A N/A N/A N/A N/A 52 b. If the data were excluded, what are the potential consequences for this study’s results? Are the valid results of this RCT important? Appraisal Criterion Reader’s Comments 10. What were the statistical findings of this study? a. When appropriate use the calculation forms below to determine these values b. Include: tests of differences With p-values and CI c. Include effect size with p-values and CI d. Include ARR/ABI and RRR/RBI with p-values and CI e. Include NNT and CI f. Other stats should be included here N/A 11. What is the meaning of these statistical N/A findings for your patient/client’s case? What does this mean to your practice? 12. Do these findings exceed a minimally N/A important difference? Was this brought up or discussed? a. If the MCID was not met, will you still use this evidence? Can you apply this valid, important evidence about an intervention in caring for your patient/client? What is the external validity? Appraisal Criterion Reader’s Comments 13. Does this intervention sound appropriate for use (available, affordable) in your clinical setting? Do you have the facilities, skill set, time, 3rd party coverage to provide this treatment? The resulting information is appropriate for any practitioner who encounters a patient with CD. The focus is more on the attitude of the practitioner and recognizing the validity of the diagnosis than the treatment. 14. Are the study subjects similar to your patient/ client? a. If not, how different? Can you use this intervention in spite of the differences? Yes. They either have conversion disorder, malingering, factitious disorder in which all would be treated similarly. 53 15. Do the potential benefits outweigh the potential risks using this intervention with your patient/client? Absolutely. All patients could benefit from a practitioner who is supportive, open-minded, and non judgmental. 16. Does the intervention fit within your patient/client’s stated values or expectations? a. If not, what will you do now? Yes the values of the patients with diagnosis want to get better. 17. Are there any threats to external validity in this study? Yes, not all treatments will work for everyone with a diagnosis of CD. The treatment is as individual as the patient. What is the bottom line? Appraisal Criterion Reader’s Comments PEDRO score N/A Summarize your findings and relate this back to clinical significance Treatment begins with the healthcare provider and their willingness to add CD as a real possibility to the differential diagnosis in a timely manner. There is mounting evidence to suggest that patients with an earlier diagnosis and treatment have a greater chance of reversal of symptoms regardless of their treatment. Goals of the diagnostic assessment should include identification of the psychosocial circumstances in which the conversion symptoms first occurred and a determination of which patients are likely to benefit from psychological exploration and insight-oriented psychotherapy as opposed to more physically based interventions and modalities such as physiotherapy and deep breathing and relaxation techniques. 54 Appendix A.5 Intervention – Evidence Appraisal Worksheet Citation: Speed, J. (1996). Behavioral management of conversion disorder: Retrospective study. Archives of Physical Medicine and Rehabilitation, 77, 147-154. Level of Evidence (Oxford scale): 2b - Retrospective case series Is the purpose and background information sufficient? Appraisal Criterion Reader’s Comments Study Purpose The purpose is clearly stated as follows: To assess whether operant behavioral treatment of conversion disorder provides effective and durable symptom resolution, and to evaluate the prognostic value of duration of symptoms, as to the time required to effect symptom resolution Stated clearly? Usually stated briefly in abstract and in greater detail in introduction. May be phrased as a question or hypothesis. A clear statement helps you determine if topic is important, relevant and of interest to you. Consider how the study can be applied to PT and/or your own situation. What is the purpose of this study? Literature Relevant background presented? A review of the literature should provide background for the study by synthesizing relevant information such as previous research and gaps in current knowledge, along with the clinical importance of the topic. Describe the justification of the need for this study Yes. I8 articles were reviewed and summarized regarding various treatment approaches for CD. The majority of studies were 1-2 subject case studies, a relatively low level of evidence. 55 Does the research design have strong internal validity? Appraisal Criterion Reader’s Comments Discuss possible threats to internal validity in the research design. Include: Assignment Attrition History Instrumentation Maturation Testing Compensatory Equalization of treatments Compensatory rivalry Statistical Regression Assignment: the first 10 patients diagnosed as having conversion disorder treated by the author in an inpatient rehab setting. All patients had CD diagnosed by a physician other than the author. All patients had gait abnormalities Attrition: Retrospective case study. All patients were evaluated into the group they were assigned. Exclusion Criteria: clearly stated but no subjects were excluded based on criteria. Treatment location: Inpatient rehab Explanation provided to the patient: A ―tests and examination established that the brain, spinal cord, nerves, and muscles were intact, and that the messages allowing normal muscle movement were being blocked.‖ It was explained that appropriate physical therapy can reestablish the normal flow of messages, and that sometimes certain stressors can make the problem worse, opening the door to psychological evaluation of the patient once the treatment regimen is underway. Patient was confined to a wheelchair at all times when not directly involved in physical therapy. There was no discussion with the patient about their abnormal gait. The conversion disorder patient must demonstrate consistent mastery of one step before proceeding to the next. Copious praise was given to the patient for mastery of each step, and no reinforcement was provided for abnormal movement or gait. 56 OT and Recreational Therapy were used for positive reinforcement. Psychological intervention began within a few days after treatment started and was directed at identifying stressors within the patient’s life and evaluating the patient’s typical way of responding to these. Attrition/Followup: All patients followed after discharge were reevaluated by the author on an outpatient basis. In 2 cases, the patient was referred to a mental health care provider in their own region for stress management and supportive counseling. No Control Group was noted. Are the results of this therapeutic trial valid? Appraisal Criterion Reader’s Comments 1. Did the investigators randomly assign subjects to treatment groups? a. If no, describe what was done b. What are the potential consequences of this assignment process for the study’s results? The first 10 patients diagnosed as having conversion disorder treated by the author in an inpatient rehab setting. All patients had CD diagnosed by a physician other than the author. All patients had gait abnormalities 2. Did the investigators know who was being assigned to which group prior to the allocation? a. If they were not blind, what are the potential consequences of this knowledge for the study’s results? 3. Were the groups similar at the start of the trial? Did they report the demographics of the study groups? a. If they were not similar – what differences existed? b. Do you consider these differences a threat to the research validity? How might the differences between groups There was no blinding. The author was the one providing the intervention, but the study was done retrospectively and included the first 10 patients to meet the criteria, thus the consequences were minimized from this element. All patients had a similar diagnosis of CD with gait abnormalities from a practitioner other than the author/provider of intervention. There were 5 men patients and 5 women patients. Age ranged from 19 to 69 years, with a mean of 32.7 years and a median of 28. Years of education ranged from 10 to 16, with a mean of 13.6. Five subjects were married and 5 were single. Duration of conversion disorder 57 affect the results of the study? symptoms ranged from 0.5 to more than I12 weeks, with a mean duration of symptoms before onset of treatment of 27.75 weeks, and a median duration of 12 weeks 4. Did the subjects know to which treatment group they were assign? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results 5. Did the investigators know to which treatment group subjects were assigned ? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results 6. Were the groups managed equally, apart from the actual experimental treatment? a. If not, what are the potential consequences of this knowledge for the study’s results? 7. Was the subject follow-up time sufficiently long to answer the question(s) posed by the research? a. If not, what are the potential consequences of this knowledge for the study’s results? 8. Did all the subjects originally enrolled complete the study? a. If not how many subjects were lost? b. What, if anything, did the authors do about this attrition? c. What are the implications of the attrition and the way it was handled with respect to the study’s findings? 9. Were all patients analyzed in the groups to which they were randomized (i.e. was there an intention to treat analysis)? a. If not, what did the authors do with the data from these subjects? The subjects were unaware they were participants in a study at the time of intervention, thus there were no consequences regarding blinding of subjects. Investigators at the time of the intervention may or may not have known they would later conduct a study on the subjects. There was only an intervention group and yes they appeared to be managed similarly. Follow up time was different for each subject. Subjects were evaluated monthly for anywhere between 7 and 36 months. All ten subjects completed the intervention, and one was lost for follow up information. All subjects were evaluated in the intervention group. No control group was utilized. 58 b. If the data were excluded, what are the potential consequences for this study’s results? Are the valid results of this RCT important? Appraisal Criterion Reader’s Comments 10. What were the statistical findings of this study? a. When appropriate use the calculation forms below to determine these values b. Include: tests of differences With p-values and CI c. Include effect size with p-values and CI d. Include ARR/ABI and RRR/RBI with p-values and CI e. Include NNT and CI f. Other stats should be included here Using a Wilcoxon signed rank test (n = 10). This demonstrates a significant change in FIM ambulation scorefrom pretreatment to posttreatment (p = .002). 11. What is the meaning of these statistical findings for your patient/client’s case? What does this mean to your practice? While behavioral modification therapy can be effective for resolution of gait abnormalities in conversion disorder, the lasting effects are not statistically significant. Furthermore, there is a significant correlation between the duration of symptoms prior to treatment and the length of time required to normalize gait. To evaluate permanence of treatment effect, n = 9 and (p = .1016) Duration of symptoms (in wks) against the amount of time (days) required to attain normal gait. There is a significantly positive correlation between these two factors (Spearman correlation coefficient 682. p = .0296). The behavioral treatment of conversion disorder symptoms clearly appears to be effective (in 100% of this study population), and treatment effect was maintained over lengthy periods of follow-up in 7 of 9 (78%) patients followed, although this did not reach statistical significance. 12. Do these findings exceed a minimally important difference? Was this brought up or discussed? a. If the MCID was not met, will you still use this evidence? MCID was not reference in article. 59 Can you apply this valid, important evidence about an intervention in caring for your patient/client? What is the external validity? Appraisal Criterion Reader’s Comments 13. Does this intervention sound appropriate for use (available, affordable) in your clinical setting? Do you have the facilities, skill set, time, 3rd party coverage to provide this treatment? Behavioral treatment of conversion disorder, based on the model described by Trieschmann, appears to be both effective and cost-effective when compared with other treatments described in the literature. 14. Are the study subjects similar to your patient/ client? a. If not, how different? Can you use this intervention in spite of the differences? Yes the study subjects are similar to my patients. They are all presenting with gait abnormalities, some are positive motor findings while others are negative motor findings. 15. Do the potential benefits outweigh the potential risks using this intervention with your patient/client? Treatment of CD is expensive in general, but not treating these subjects is not a reasonable option seeing that many of them are functionally impaired limiting their ability to work and function in society. What studies are showing is that treating the physical symptoms while treating the psychological symptoms may actually decrease overall health care costs as well as assist the subject in returning to a more functional and contributory role in society. 16. Does the intervention fit within your patient/client’s stated values or expectations? a. If not, what will you do now? Yes as patients with conversion disorder do typically want to get better, and preferably without the stigma associated with purely psychological treatment. 17. Are there any threats to external validity in this study? Not all patients diagnosed with conversion disorder actually have conversion disorder. However, I believe that treating similar diagnoses, to include malingering, with similar treatment behavioral modification techniques can yield similar results. 60 What is the bottom line? Appraisal Criterion Reader’s Comments PEDRO score (see scoring at end of form) N/A Summarize your findings and relate this back to clinical significance Limitations of this study include its retrospective, non-blinded design, and the fact that different observers collected FIM ambulation scores. Followup times were unable to be standardized and sample size and lack of a control group are obvious weaknesses. However, the conclusions drawn from the data appear valid. At least initially, physical therapy intervention using behavior modification techniques, can greatly improve functional independence and normalize gait patterns in a relatively short inpatient rehabilitation stay when paired with a multidisciplinary approach. 61 Appendix A.6 Systematic Review – Evidence Appraisal Worksheet Citation: Sumathipala , A. (2007). What is the evidence for the efficacy of treatments for somatoform disorders? a critical review of previous intervention studies. Psychosomatic Medicine, 69, 889-900. Level of Evidence (Oxford scale): 1A – SR of SR’s and RCT’s Does the design follow the Cochrane method? Appraisal Criterion Reader’s Comments Step 1 – formulating the question The term Medically Unexplained Symptom (MUS) and patients presenting with MUS, to include all functional problems rather than just the sub-groups who met the operational criteria for somatization disorder, conversion disorder, or symptoms syndromes were identified for review. This review attempted to answer the question, what is the highest level of evidence available for the efficacy of pharmaceutical and non-pharmaceutical interventions for patients with MUS and where have these studies been carried out? The aim was to identify the gaps and then to report any recent advances made since the last systematic review. • • Do the authors identify the focus of the review A clearly defined question should specify the types of: • people (participants), • interventions or exposures, • outcomes that are of interest • studies that are relevant to answering the question Step 2 – locating studies Should identify ALL relevant literature Did they include multiple databases? Was the search strategy defined and include: o Bibliographic databases used as well as hand searching o Terms (key words and index terms) o Citation searching: reference lists o Contact with ‘experts’ to identify ‘grey’ literature (body of materials that cannot be found easily through conventional channels such as publishers) o Sources for ‘grey literature’ An extensive literature review was conducted using electronic databases to include: Cochrane library databases (up to 2007), Medline (1966 –2007), PsychINFO (1974 –2006), and EMBASE (1980 – 2007). The following key words were used first individually in the abstract, key words, and title: ―medically unexplained symptoms,‖ ―somatisation,‖ ―somatization,‖ then in combination with psychological therapies, cognitive behavior therapy, pharmacological therapies, management, therapy, drug therapy, and antidepressants, and ―somatoform disorders‖. To ensure a comprehensive review, search for literature was supplemented by examining the reference lists of the papers generated from the original searches. 62 Part 3:Critical Appraisal/Criteria for Inclusion • Were criteria for selection specified? • Did more than one author assess the relevance of each report • Were decisions concerning relevance described; completed by non-experts, or both? • Did the people assessing the relevance of studies know the names of the authors, institutions, journal of publication and results when they apply the inclusion criteria? Or is it blind? Abstracts eligible for inclusion were systematic reviews or randomized trials of a psychological, pharmacological, or any other type of intervention involving an adult, with patients defined as medically unexplained symptoms, unexplained symptoms, somatoform disorders, somatisation, somatization, functional somatic symptoms, and the abstract was written in the English language. All abstracts selected were checked for duplications, which, if found were excluded. Systematic reviews or RCTs exclusively on symptom syndromes, such as irritable bowel syndrome, chronic fatigue syndrome (CFS), and fibromyalgia, were excluded. Systematic reviews of MUS incorporating symptom syndromes, however, were included. Papers focusing on children and adolescents were also excluded. Literature review, this section does not apply Part 3 – Critically appraise for bias: • Selection – • Were the groups in the study selected differently? • Random? Concealed? • Performance• Did the groups in the study receive different treatment? • Was there blinding? • Attrition – • Were the groups similar at the end of the study? • Account for drop outs? • Detection – • Did the study selectively report the results? • Is there missing data? Part 4 – Collection of the data Was a collection data form used and is it included? Are the studies coded and is the data coding easy to follow? Were studies identified that were excluded & did they give reasons why (i.e., which criteria they failed). Charts are provided for search results, with explanation of which articles were selected for analysis. 63 Are the results of this SR valid? Appraisal Criterion Reader’s Comments 18. Is this a SR of randomized trials? Did they limit this to high quality studies at the top of the hierarchies a. If not, what types of studies were included? b. What are the potential consequences of including these studies for this review’s results? 19. Did this study follow the Cochrane methods selection process and did it identify all relevant trials? a. If not, what are the consequences for this review’s results? Yes, only SR and RCT’s were included. This increases the quality of the study and provides more reliable and valid conclusions drawn from results. All relevant trails were identified, making this an exhaustive review of the current research. Even articles that were referenced in the selected articles were reviewed for relevance and possible inclusion to confirm this. 20. Do the methods describe the processes and tools used to assess the quality of individual studies? a. If not, what are the consequences for this review’s results? 21. What was the quality of the individual studies included? Were the results consistent from study to study? Did the investigators provide details about the research validity or quality of the studies included in review? 22. Did the investigators address publication bias Are the valid results of this SR important? Yes. Methods and charts include analysis of number of subjects, treatment conditions vs control conditions, and reports and conclusions drawn by authors. Appraisal Criterion Reader’s Comments Each study was ranked either as Level 1 evidence or Level 2 evidence and analyzed appropriately in the group it was ranked. No this was not specifically addressed. 23. Were the results homogenous from study to study? a. If not, what are the consequences for this review’s results? Results between studies varied but generally trended towards at least short term efficacy of cognitive behavioral therapy and use of antidepressants for symptoms of MUS. 24. If the paper is a meta-analysis did they report the statistical results? Did they include a forest plat? What other statistics do they include? Are there CIs? Not a meta-analysis. 64 25. From the findings, is it apparent what the No. While the finding suggests significant decrease cumulative weight of the evidence is? in medical cost pending alternative treatments for MUS, the general conclusion of the paper is that more pragmatic controlled trials are necessary to be significant. Can you apply this valid, important evidence from this SR in caring for your patient/client? What is the external validity? Appraisal Criterion Reader’s Comments 26. Is your patient different from those in this SR? No. My patient demonstrated MUS, and multiple very expensive interactions with the health care system to try and decipher what the issue was. While he was on anti-depressants he was not covered by insurance to attempt CBT in the physical therapy setting. Physical therapy is not specifically referenced in this paper. 27. Is the treatment feasible in your setting? Do you have the facilities, skill set, time, 3rd party coverage to provide this treatment? 28. Does the intervention fit within your patient/client’s stated values or expectations? a. If not, what will you do now? Yes, CBT can be implemented at low cost, at low risk to the patient. Yes. What is the bottom line? Appraisal Criterion Reader’s Comments Summarize your findings and relate this back to clinical significance The review reveals that two types of interventions, antidepressant medication and CBT are supported by level I evidence as benefiting patients with MUS. There is more level I evidence for CBT compared with other approaches and the evidence is increasing. CBT seems to be effective in the reduction of a wide range of physical symptoms and associated mood disturbance, as well as in producing improvements in overall physical and social functioning. Antidepressants are moderately 65 effective for MUS and effect sizes are homogenous across functional syndromes but are associated withdrawal symptoms and side effects. There are currently no trials comparing antidepressants with CBT. 66 Appendix A.7 Intervention – Evidence Appraisal Worksheet Citation: Tocchio, S. (2009). Treatment of conversion disorder: A clinical and holistic approach. Journal of Psychosocial Nursing, 47(8), 42-47. Level of Evidence (Oxford scale): 5 – Case Report Is the purpose and background information sufficient? Appraisal Criterion Reader’s Comments Study Purpose The intent of this article is to encourage others by describing the success that psychiatry and alternative medicine can offer to patients with conversion disorder and to emphasize the value of nursing as a part of that team. Stated clearly? Usually stated briefly in abstract and in greater detail in introduction. May be phrased as a question or hypothesis. A clear statement helps you determine if topic is important, relevant and of interest to you. Consider how the study can be applied to PT and/or your own situation. What is the purpose of this study? Literature Yes. The history of the diagnosis, and prevalence was discussed. Treatment options by psychologist’s tends to be secondary with initial treatments provided by neurologists or other medical professionals were also described. Relevant background presented? A review of the literature should provide background for the study by synthesizing relevant information such as previous research and gaps in current knowledge, along with the clinical importance of the topic. Describe the justification of the need for this study 67 Does the research design have strong internal validity? Appraisal Criterion Reader’s Comments N/A. Single subject case study. Discuss possible threats to internal validity in the research design. Include: Assignment Attrition History Instrumentation Maturation Testing Compensatory Equalization of treatments Compensatory rivalry Statistical Regression Are the results of this therapeutic trial valid? Appraisal Criterion Reader’s Comments 1. Did the investigators randomly assign subjects to treatment groups? a. If no, describe what was done b. What are the potential consequences of this assignment process for the study’s results? 2. Did the investigators know who was being assigned to which group prior to the allocation? a. If they were not blind, what are the potential consequences of this knowledge for the study’s results? 3. Were the groups similar at the start of the trial? Did they report the demographics of the study groups? a. If they were not similar – what differences existed? b. Do you consider these differences a threat to the N/A. Single subject case study. N/A. Single subject case study. N/A. Single subject case study. 68 4. 5. 6. 7. 8. 9. research validity? How might the differences between groups affect the results of the study? Did the subjects know to which treatment group they were assign? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results Did the investigators know to which treatment group subjects were assigned ? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results Were the groups managed equally, apart from the actual experimental treatment? a. If not, what are the potential consequences of this knowledge for the study’s results? Was the subject follow-up time sufficiently long to answer the question(s) posed by the research? a. If not, what are the potential consequences of this knowledge for the study’s results? Did all the subjects originally enrolled complete the study? a. If not how many subjects were lost? b. What, if anything, did the authors do about this attrition? c. What are the implications of the attrition and the way it was handled with respect to the study’s findings? Were all patients analyzed in the groups to which they were randomized (i.e. was there an intention to treat analysis)? a. If not, what did the authors do with the data from these subjects? b. If the data were excluded, what are the potential consequences for this study’s results? Yes, single subject case study. Yes, single subject case study.. N/A. Single subject case study. There was no follow up, thus one cannot conclude if the treatment was effective in the long term. The only subject involved completed the study. N/A. Single subject case study. 69 Are the valid results of this RCT important? Appraisal Criterion Reader’s Comments 10. What were the statistical findings of this study? a. When appropriate use the calculation forms below to determine these values b. Include: tests of differences With p-values and CI c. Include effect size with p-values and CI d. Include ARR/ABI and RRR/RBI with p-values and CI e. Include NNT and CI f. Other stats should be included here There is no statistical analysis completed for this case study. 11. What is the meaning of these statistical findings for your patient/client’s case? What does this mean to your practice? 12. Do these findings exceed a minimally important difference? Was this brought up or discussed? a. If the MCID was not met, will you still use this evidence? N/A N/A Can you apply this valid, important evidence about an intervention in caring for your patient/client? What is the external validity? Appraisal Criterion Reader’s Comments 13. Does this intervention sound appropriate for use (available, affordable) in your clinical setting? Do you have the facilities, skill set, time, 3rd party coverage to provide this treatment? Biofeedback and guided imagery seems relevant to the PT setting, and may be justifiable to insurance. Biofeedback mechanisms can be purchased for home use for continued assistance outside the skilled therapy session. 14. Are the study subjects similar to your patient/ client? a. If not, how different? Can you use this intervention in spite of the differences? Yes. The patient has diagnosed conversion disorder. Her symptoms are more associated with negative motor findings, while my patient had positive motor findings, but the root cause is the same. 70 15. Do the potential benefits outweigh the potential risks using this intervention with your patient/client? Yes. The general trend of the health care system in the US is leaning more towards holistic approaches, and in the diagnosis of CD, and other MUS, standard medical treatment may be less effective than holistic treatments. Risks to the patient are low, in this case the costs are high due to lack of insurance coverage. 16. Does the intervention fit within your patient/client’s stated values or expectations? a. If not, what will you do now? Yes, the patients in general are seeking symptom resolution, which was demonstrated through this study. 17. Are there any threats to external validity in this study? Yes, it is only one patient. The care was paid for out of pocket by the parents of the patient, and not covered by insurance. What is the bottom line? Appraisal Criterion Reader’s Comments PEDRO score (see scoring at end of form) N/A Summarize your findings and relate this back to clinical significance Every patient with diagnosed Conversion Disorder will present with various symptoms and respond to treatments to differently. This particular patient rejected behavioral modification techniques and positive verbal encouragement stating that it increased her level of stress and initiated her symptoms more frequently. It is important to consider these variations and keep an open mind to a variety of treatment options to include holistic therapies. This study is limited as it is a case report with limited external validity. Physical therapy was not specifically mentioned as an element of care in the disciplinary team, however, biofeedback as a tool to assist in treatment of Conversion Disorder, by a physical therapist, is applicable. 71 Appendix A.8 Intervention – Evidence Appraisal Worksheet Citation (use AMA or APA format): Voon, V., Brezing, C., Gallea, C., Ameli, R., Roelofs, K., LaFrance Jr, W., & Hallett, M. (2010). Emotional stimuli and motor conversion disorder. Brain, 133, 1526-1536. Level of Evidence (Oxford scale): 2b – Individual cohort study (including low quality RCT; <80% f/up) Is the purpose and background information sufficient? Appraisal Criterion Reader’s Comments Study Purpose Purpose is not clearly stated, however it can be deduced that their intention was to ―We sought to investigate the relationship between affect or arousal and conversion disorder with positive motor symptoms (herein referred to as motor conversion disorder) by investigating amygdala activity in association with viewing affective stimuli in a large patient sample size.‖ Stated clearly? Usually stated briefly in abstract and in greater detail in introduction. May be phrased as a question or hypothesis. A clear statement helps you determine if topic is important, relevant and of interest to you. Consider how the study can be applied to PT and/or your own situation. What is the purpose of this study? Literature Relevant background presented? Relevant background evidence is presented. Functional imaging studies have focused on conversion paralysis or the absence of movement. In this study, they focused on conversion disorder with positive motor symptoms such as tremor, dystonia, chorea, tics and gait disorders rather than conversion paralysis. A review of the literature should provide background for the study by synthesizing relevant information such as previous research and gaps in current knowledge, along with the clinical importance of the topic. Describe the justification of the need for this study 72 Does the research design have strong internal validity? Appraisal Criterion Reader’s Comments Clear inclusion/exclusion and control group matching noted below, and study was approved by the National Discuss possible threats to internal validity in the research design. Include: Assignment Attrition History Instrumentation Maturation Testing Compensatory Equalization of treatments Compensatory rivalry Statistical Regression Institutes of Health Institutional Review Board and all subjects signed informed consent: Inclusion criteria for patients with motor conversion disorder Included diagnostic confirmation by at least two neurologists and one psychiatrist, no movement symptoms at rest for the imaging study, movement symptoms not affecting the head or neck, no history of traumatic brain injury and not on antidepressants and 19 years of age or older. Exclusion criteria including having a serious medical or neurological illness, current major depression, panic disorder, post-traumatic stress disorder, substance abuse or other major affective or psychotic disorders, being on antidepressants and contraindications for MRI. Age- (+5 years) and gender-matched healthy volunteers were recruited from the National Institutes of Health healthy volunteer database. Are the results of this therapeutic trial valid? Appraisal Criterion Reader’s Comments 18. Did the investigators randomly assign subjects to treatment groups? a. If no, describe what was done b. What are the potential consequences of this assignment process for the study’s results? No. Groups were separated into those with the diagnosis and those without diagnosis, and all were provided the same stimulus and imaging protocol. 73 19. Did the investigators know who was being assigned to which group prior to the allocation? a. If they were not blind, what are the potential consequences of this knowledge for the study’s results? 20. Were the groups similar at the start of the trial? Did they report the demographics of the study groups? a. If they were not similar – what differences existed? b. Do you consider these differences a threat to the research validity? How might the differences between groups affect the results of the study? 21. Did the subjects know to which treatment group they were assign? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results Allocation was not concealed from those collecting data or analyzing findings. 22. Did the investigators know to which treatment group subjects were assigned ? a. If yes, what are the potential consequences of the subjects’ knowledge for this study’s results 23. Were the groups managed equally, apart from the actual experimental treatment? a. If not, what are the potential consequences of this knowledge for the study’s results? 24. Was the subject follow-up time sufficiently long to answer the question(s) posed by the research? a. If not, what are the potential consequences of this knowledge for the study’s results? 25. Did all the subjects originally enrolled complete the study? a. If not how many subjects were lost? b. What, if anything, did the authors Blinding was not used in this study, and it could very well affect the outcome analysis based on preconceived notions regarding the diagnosis. Groups were similar, age and gender matched, all right handed individuals except 1 left handed subject with CD, 16 subjects in each group. Each group received the same treatment and they were aware of their diagnosis or lack there of. I do not believe this to be a threat to internal validity due to the design of the study being a comparison of diagnosis vs no diagnosis. Groups were managed equally, receiving the same intervention and the same analysis between groups. No follow up was completed. Treating the conversion disorder and repeating the MRI testing and questionnaires would be a great way of taking this research to the next level. All subjects originally enrolled in the study completed the study. 74 do about this attrition? c. What are the implications of the attrition and the way it was handled with respect to the study’s findings? 26. Were all patients analyzed in the groups to which they were randomized (i.e. was there an intention to treat analysis)? a. If not, what did the authors do with the data from these subjects? b. If the data were excluded, what are the potential consequences for this study’s results? Are the valid results of this RCT important? All subjects were analyzed in the groups they were assigned. Appraisal Criterion Reader’s Comments 27. What were the statistical findings of this study? a. When appropriate use the calculation forms below to determine these values b. Include: tests of differences With p-values and CI c. Include effect size with p-values and CI d. Include ARR/ABI and RRR/RBI with p-values and CI e. Include NNT and CI f. Other stats should be included here 28. What is the meaning of these statistical findings for your patient/client’s case? What does this mean to your practice? Behavioral effects: There was no difference in reaction time between patients with motor conversion disorder and healthy volunteers. Imaging effects: Patients with motor conversion disorder had greater right amygdala activity compared to healthy volunteers in healthy and fearful vs. neutral and rest. There was no difference in amygdala activity when the neutral condition was compared between patients with motor conversion disorder and healthy volunteers. Results may show that in patients with CD either a role for generalized increased arousal or alternatively, a failure of adaptation of the attentional process in evaluating salience relevance with repeated presentations. Arousal on the amygdala activity may influence motor symptoms either through a general effect on initiation of the motor conversion symptom or possibly through failure of inhibition of the motor conversion symptom. This suggests that our attitude and emotions present when treating patients may affect their symptoms and outcomes. 75 29. Do these findings exceed a minimally The MCID was not discussed, but this information is important difference? Was this brought still relevant to my practice as general information up or discussed? on CD and the brain function in response to stimuli. a. If the MCID was not met, will you still use this evidence? Can you apply this valid, important evidence about an intervention in caring for your patient/client? What is the external validity? Appraisal Criterion Reader’s Comments 30. Does this intervention sound appropriate for use (available, affordable) in your clinical setting? Do you have the facilities, skill set, time, 3rd party coverage to provide this treatment? This intervention is not appropriate for the scope of physical therapy. It may later be useful as a means of diagnosis of CD, or developing biological or psychological treatments targeting arousal. 31. Are the study subjects similar to your patient/ client? a. If not, how different? Can you use this intervention in spite of the differences? The study subjects are similar to my patient population all subjects with CD presented with positive motor symptoms mimicking neurological conditions. 32. Do the potential benefits outweigh the potential risks using this intervention with your patient/client? Utilizing varying arousal levels for interaction with patients based on their response as part of a behavioral management approach to therapy is cost effective, and would require little to no extra time to implement. 33. Does the intervention fit within your patient/client’s stated values or expectations? a. If not, what will you do now? The intervention does fit with the patients stated values as it may assist in decreasing their symptoms. 34. Are there any threats to external validity in this study? Conversion disorder also manifests as negative motor symptoms. However, the effects of arousal stimulus on the brain are probably similar based on this study. 76 What is the bottom line? Appraisal Criterion Reader’s Comments PEDRO score (see scoring at end of form) 4/10. Blinding of subjects is not applicable in this design, but blinding researchers would be beneficial. Summarize your findings and relate this back to clinical significance Motor conversion disorder is characterized by greater amygdala activity to arousal and potential impairments in habituation to arousing stimuli. These study results may not only identify functional MRI as a form of secondary confirmation of conversion disorder, but also encourage further research towards biological or psychological treatments targeting arousal as an alternative form of treatment for conversion disorder. 77 Pedro Scoring System for Voon et. al.: x x x x x x x x x x x Adapted from : Jewell, D. Guide to Evidence Based Physical Therapy Practice. Jones and Bartlett Publishers, Sudbury, MA 2008 78 Appendix B. Oxford Centre For Evidence Based Medicine 79 Appendix C. Physiotherapy Evidence Database 80