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Oral health status and behaviour of Greek patients with cleft lip and palate V. Parapanisiou *, S. Gizani *, M. Makou **, L. Papagiannoulis * Depts. of *Paediatric Dentistry and ** Orthodontics, Dental School, University of Athens, Greece. Abstract Aim: This was to record the oral health profile and more specifically the prevalence of carious and hypoplastic lesions in children and adolescents with cleft lip and palate (CLP). Study Design and Methods: The study group consisted of 41 children and adolescents (4-18 years-old) with CLP group while a similar number of non-cleft persons (HLP) matched for sex, age and orthodontic treatment (75.6%), was used as the control group. Information regarding the oral health habits, medical and dental history were collected using a questionnaire. Stimulated saliva was collected to evaluate the flow rate and buffering capacity as well as the levels of mutans streptococci and lactobacilli using the chair-test CRT® (Ivoclar –Vivadent). Oral hygiene (OH) was assessed using the index of Silness and Loe [1964]. The prevalence of initial/white spot and cavitated carious lesions as well as hypoplasia was evaluated based on the criteria by ICDAS [2005] and Nyvad et al [2008] as well as Koch et al [1987] respectively. The statistical analysis was carried out using the t-test and the chi- square test. RESULTS: Approximately half of the CLP and HLP subjects were brushing their teeth 2 x per day. Both groups reported an average of 3 main and 2 snack meals per day. Levels of mutans streptococci and lactobacilli as well as the quality of the saliva were similar for both groups. The plaque index score was significantly higher in the CLP than in the control group (p=0.0003). The prevalence of cavitated carious lesions was similar in both groups but that of the initial/white spot lesions, especially at the area of maxillary incisors, was higher in the CLP group (85%, p=0.000). Conclusions: The increased numbers of initial/ white spot lesions combined with poor OH found in the CLP group predispose for an increased risk of further development to carious cavitated lesions. Taking into consideration that the majority of those patients were under orthodontic treatment, the application of an intensive individualized oral health preventive program, focused on remineralisation of the initial caries, is imperative. Introduction Cleft lip and palate (CLP) is one of the most common congenital malformations. Epidemiological studies have shown great variation in its occurrence and its prevalence is reported to range from 0.91 to 2.69 per 1,000 in Caucasians and from 0.85 to 2.68 per 1,000 in Japanese [Vanderas, 1987]. An epidemiologic study in Greece showed that the total prevalence for the years 1985-1988 and 1989 was estimated to be 0.47/1000 live births for CLP and 0.34/1,000 live births for cleft palate only [Nasika, 1997]. Most studies have shown higher caries prevalence [Johnsen and Dixon, 1984; Dahllof et al, 1989; Bokhout et al., 1996a] and poorer oral hygiene (OH) in CLP children than in healthy children of the same age while other studies have not been able to confirm these findings [Al-Wahadi et al., 2005; Bokhout et al., 1996b]. These contradictory results have been attributed to factors such as the small size and the large age-range of the sample, the lack of control group and the absence of differentiation between the various types of cleft [Hasslof and Twetman, 2007]. The aim of this study was to a) register the oral hygiene and dietary habits, b) to assess the OH status including caries and dental anomalies and c) to evaluate the levels of cariogenic micro organisms and the quality of saliva in Greek patients with CLP. The null hypothesis was that OH status of CLP children and adolescents does not differ from that of the HLP group. Materials and Methods Subjects. The cleft group (CLP) consisted of 41 children with CLP (23 boys and 18 girls) aged 4-18 years (mean age 10.54, SD±=3.37 years). Those children with multiple abnormalities or recognized syndromes were excluded. The study group consisted of 26 children and adolescents with unilateral CLP, 10 with bilateral CLP and 5 with cleft palate; 75.6% of the patients were under orthodontic treatment. The control group (HLP) consisted of 41 healthy children and adolescents with mean age of 10.7 years (SD±3.03 years) matched for age, gender and orthodontic treatment. Both groups were selected from the patients of the Postgraduate Paediatric and Orthodontic Dental Clinic of the University of Athens. Questionnaire. Using a questionnaire completed by the parents/patients, information was obtained concerning the OH habits (tooth brushing frequency, parental help during toothbrushing, use of dental floss, age of the first dental visit, frequency of dental attendance), the fluoride exposure (application of topical fluoride and the use of fluoride supplements, such as fluoride tablets) as well as the dietary habits Key words: cleft lip; cleft palate; oral health; caries; children and adolescents. Postal address: Prof. L. Papagiannoulis. Dept of Paediatric Dentistry, University of Athens, Thivon 2, Goudi, Athens, 11527,Greece. Email: [email protected] 85 European Archives of Paediatric Dentistry // 10 (2). 2009 Parapanisiou et al. (frequency of daily consumption of juices and refreshments, the number of main meals and snacks as well as the number of meals containing sugars per day) of the study participants. Saliva evaluation and assessment of cariogenic microbiota. Stimulated saliva was collected after chewing a paraffin gum for 5 minutes and expectorating into a cup. The salivary secretion rate was evaluated in ml/min. The number of lactobacilli (LB) and mutans streptococci (MS) in saliva was detected using the CRT® chair-side tests (Ivoclar-Vivadent). The buffering capacity of the saliva was determined using the CRT® buffer test (Ivoclar-Vivadent). Evaluation of oral health. All the children had a clinical examination by two trained and calibrated paediatric dentists. The intra- and inter-examiner reliability tests for caries, initial/ white spot lesions and hypoplasia were performed prior to the study and showed kappa values greater than 0.8. OH was assessed using the plaque index (PI) of Silness and Loe [1964] on the six Ramfjord teeth (16,12,24,36,32,44 proximal, buccal, lingual) with the use of a disclosing agent (score 0=no dental plaque in the gingiva area, score 1= a film of dental plaque adhering to the free gingival margin (only upon probing), score 2=moderate accumulation of dental plaque at the gingival margin (seen by naked eye), score 3=abundant dental plaque in the gingival margin). Dental caries was assessed after professional cleaning of the tooth surfaces and both cavitated and initial/white spot lesions were registered. Cavitated carious lesions were diagnosed after visual examination using a blunt probe and a dental mirror according to the criteria by Koch et al. [1967]: score 1= the explorer sticks to the tooth surface and demands effort in order to be removed, score 2= loss of dental tissue with cavitation. The dmf/DMF indices were used to express caries experience [Klein, 1939]. The evaluation of the caries was also based on bite-wings radiographs. Initial/white spot lesions were defined as opacities of white, or brownish colour with oval or round shape following the gingival margin with decreased enamel translucency and clearly defined from the adjacent enamel. They usually appeared on single teeth and predominantly on the incisal two thirds of the crown. [ICDAS, 2005; Nyvad et al., 2008]. Hypoplasia was defined as when a tooth surface was determined as showing that its colour was white, yellow or brown and its surface was disintegrated, abraded, or even having an atypical restoration (extensive, irregular and atypical restoration replacing broken and hypomineralized enamel) [Koch et al., 1987]. Anomalies of the teeth and evaluation of the occlusion was noted when there was presence of crowding, and of posterior or anterior crossbite as well as the type of malocclusion were recorded. Developmental anomalies related to the number, size and morphology of the teeth were clinically and radiographically evaluated by panoramic radiograph. 86 European Archives of Paediatric Dentistry // 10 (2). 2009 Statistical analysis. Descriptive statistics, including mean value and standard deviation of each clinical entity, was carried out for all the parameters examined. The level of significance was set at p<0.05. The t-test and Chi-Square test were used to examine the difference between the two groups concerning all the parameters registered. The same tests were also used to investigate the differences in the OH condition and microbiological parameters between children from the CLP group and HLP group that were undergoing orthodontic treatment. Results Questionnaire. Based on the questionnaire, toothbrushing frequency was found to be similar in both groups (cleft and control) with approximately half of the children brushing their teeth 2 x per day (Figure 1a). From the cleft group, 53.7% of the parents reported that they supervised their children’s toothbrushing on a daily basis while in the control group, 39% of the parents did so (Figure 1b). Significantly (p<0.05) more patients with CLP were taking fluoride supplements (29%) than the ones in the control group (5%). The majority of patients from both groups had had at least one professional fluoride application in the last 12 months (80.4% of the CLP and 90.2% of HLP group, p>0.05). Data on the frequency of daily consumption of juices and refreshments showed that few patients from both groups consumed drinks more than 3 times daily (7.3% and 4.9% of CLP and HLP respectively, p>0.05) while 61% from both groups consumed drinks 1 to 3 times daily. Children of both groups had an average of 3 main and 2 snack meals per day, while, on an average, one and a half of those meals contained sugars. Saliva evaluation and assessment of cariogenic microbiota. More than 70% of children from both CLP and HLP had high levels of mutans streptococci and lactobacilli (>105 cfu/ml). Normal salivary flow rate (>0.7ml/min) was found in 55.2% of CLP children and 65.9% of HLP children. High buffering capacity was found in 91.9% of children with CLP and 92.6% of HLP children. Differences were not statistically significant (p>0.05). Patients from both CLP and HLP group being under orthodontic treatment had high levels of cariogenic bacteria (Table 1). Oral health evaluation. The plaque index of the total dentition was significantly higher in the CLP than in the control group (CLP group, mean score: 1.03, SD: 0.35 vs HLP group, mean score: 0.76, SD±0.29, p=0.0003) and the same applied for the teeth adjacent to the cleft (CLP group, mean score: 1.11, SD±0.52 vs HLP group, mean score: 0.82, SD±0.40, p=0.0027). The tendency was similar when the factor of orthodontic treatment was taken into consideration. CLP subjects under orthodontic treatment had worse OH than those in the HLP group (Table 1). No significant differences were found in the prevalence of cavitated carious lesions between patients with cleft and without. The mean dmfs score was found to be 7.24 for the Oral health of patients with cleft lip and palate Figure 1a Figure 1b Figure 1. Bar graphs showing percentage of: a. Toothbrushing frequency in the CLP and the HLP group; b. Parental supervision during toothbrushing in the CLP and the HLP group. Figure 2. Caries experience in the primary dentition in the CLP and HLP groups. 87 European Archives of Paediatric Dentistry // 10 (2). 2009 Parapanisiou et al. CLP and 8.38 for the HLP group (Figure 2). For both groups, the filled surfaces component was the largest of the dmfs index mean score. A similar picture was seen for the permanent dentition (CLP: mean score: DMFS 3.00 and FS 2.39 vs HLP: mean score: DMFS 3.40 and FS 2.05). The situation was also similar between CLP and HLP subjects under orthodontic treatment (Table 1). The majority of initial/white spot lesions in the permanent dentition were found in the CLP group (85%, p=0.000). Most of patients in the CLP group had 1 to 3 initial/white spot lesions, mainly on the maxillary front teeth (Table 2). One should take into consideration that most of children in CLP group were under orthodontic treatment. The percentage of children with enamel hypoplasia was higher in the CLP group than in the HLP group (29.3% versus 17% respectively). However this difference was not statistically significant (p>0.05). Supernumerary teeth were not found in the control group while 9.8% of children from CLP group had at least one supernumerary tooth (p<0.05). Rotated and missing teeth were found significantly more often in the CLP group than in the HLP group (68.3% and 43.9% vs 29.3% and 5% respectively, p=0.000). Anterior and posterior crossbites were found more often in the CLP group than in the HLP group (58.5% and 39% vs 9.8% and 0%, respectively, p<0.001) Discussion This was a comparative cross-sectional study of children with and without CLP matched for sex, age and orthodontic treatment. The results from the questionnaire showed that the majority of the study subjects with and without CLP brushed their teeth at least once a day without parental supervision in most cases. This is in agreement with the results of the study by Dahllof et al. [1989], which is the only study, as far as we know, that compared the OH habits between CLP and HLP children and adolescents. Table 1. Oral health status in CLP and HLP in a group of Greek children going under orthodontic treatment. CLP (mean value) HLP (mean value) P1I * 1.04 0.80 PlIs ** 1.19 0.85 Dmft 3.00 4.16 DMFT 2.61 1.25 SM >105 cfu/ml 78.57% 85.7% LB >10 cfu/ml 75% 64.2% 5 CLP: patients with cleft; HLP: healthy patients; PlI: plaque index on Ramfjord teeth, respectively; PlIs: plaque index on teeth situated near the cleft, respectively; dmft: caries index in the deciduous dentition; DMFT: caries index in the permanent dentition; MS: levels of mutans streptococci; LB: levels of lactobacilli; *p=0.004; **p=0.000 88 European Archives of Paediatric Dentistry // 10 (2). 2009 Table 2. Distribution of initial/white spot lesions in the CLP and HLP in a group of Greek children under going orthodontic treatment. Number of initial/white spot lesions 0 1 2 3 4 5 6 8 10 CLP 17 7 4 5 3 2 1 1 1 HLP 35 3 - 2 1 - - - - CLP: patients with cleft; HLP: healthy patients Regarding the use of fluoride supplements, the CLP group received fluoride tablets more often than that of HLP group (29.2% vs 4.8%). Although this difference was statistically significant, this result should be evaluated with caution as this finding was based on a reported behaviour. In assessing the prevalence of cavitated carious lesions in both the CLP and the HLP group, the findings in our study are in agreement with those of other studies [Lauterstein and Mendelsohn, 1964; Lucas et al., 2000]. In a recent systematic review, Hasslof and Twetman [2007] concluded that the literature has failed to prove that there is difference in the caries status between patients with and without CLP, up to now. In our study, caries diagnosis was based on both clinical and radiographic examination. While no differences were found in the prevalence of cavitated carious lesions, significantly more initial/white spot lesions were found in CLP than HLP group (85% vs 15%). Our finding is in agreement with the study by Bokhout et al. [1996]. It seems that patients with CLP may be at greater risk for progression of initial/ white spot lesions to cavitated caries. Primarily the poor OH in CLP patients might explain this finding. Possible reasons for the poorer OH hygiene in CLP group are the following: the difficulty in tooth cleaning because of the presence of residual scar tissue as a result of the multiple surgical procedures carried out at the cleft region; the lack of interest for oral hygiene due to many other health problems such as otitis media, difficulty in speech and the fear that children often have when they brush their teeth at the cleft area. All the above factors make the importance of the application of individualized preventive oral health programs in CLP patients, imperative. The majority of children from CLP and HLP group had high levels of mutans streptococci and lactobacilli. This can be partially explained by the fact that both groups had poor OH habits and similar caries experience as they were all patients of the paediatric dental clinic where mostly high caries risk patients are seeking oral health care. Concerning the dental anomalies, supernumerary, missing and rotated teeth were found to be significantly more frequent among the children with cleft (p<0.05). Furthermore Oral health of patients with cleft lip and palate the prevalence of enamel hypoplasia was higher in children with CLP than those without, but the difference was not statistically significant. These findings are in agreement with the literature [Dahllof et al., 1989; Vichi and Franchi, 1995; Slayton et al., 2003; Aizenbud et al., 2005]. The differences in the prevalence of the dental anomalies in our study, as well as in the literature, may be due to differences in the size and the method of sampling plus the fact that most of the studies did not have a control group [Aizenbud et al., 2005]. The design of the present study took into consideration the criteria introduced by Twetman and Hasslof [2007]. However due to the small size and the wide age-range of the sample, it should be considered as a pilot study. Nevertheless this research is the only study of its kind that has ever been carried out in Greece. Conclusion The increased numbers of initial/white spot lesions combined with poor OH found in a Greek group of children with CLP predisposes to an increased risk for further development of carious cavitated lesions. As the majority of CLP patients in the study were under orthodontic treatment, the application of an intensive individualized OH preventive program focused on the improvement of OH and remineralisation of initial/white spot carious lesions is imperative. Acknowledgements Approval for the study was obtained from the Ethics Committee of the Dental School of the University of Athens; a written consent from all the participants/ parents was obtained. References Aizenbud D, Camasuvi S, Peled M, Brin I Congenitally missing teeth in the Israeli cleft population. Cleft Palate - Craniofacial J 2005;42:314-317. Al –Wahadni A, Alhaija EA, Al-Omari MA Oral disease status of a sample of Jordanian people ages 10 to 28 with cleft lip and palate. Cleft Palate Craniofacial J 2005;42:304-308. Bokhout B, Hofman FXWM, van Limbeek J, Kramer GJC, Prahl-Andersen B Increased caries prevalence in 2.5 –year-old-children with cleft lip and/or palate. Eur J Oral Sci 1996;104:518-22. 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Koch G, Hallonsten A-L, Ludvigsson N, et al. Epidemiologic study of idiopathic enamel hypomineralization in permanent teeth of Swedish children. Community Dent Oral Epidemiol 1987;15:279-285. Koch G. Effect of sodium fluoride in dentifrice and mouthwash on incidence of dental caries in schoolchildren. Odontol Revy 1967;18(Suppl 12). Lauterstein AM, Mendelsohn M. An analysis of the caries experience of 285 cleft palate children. Cleft Palate Craniofac J 1964;1:314-319. Lucas VS, Gupta R, Ololade O, Gelbier M, Roberts GJ. Dental health indices and caries associated microflora in children with unilateral cleft and palate. Cleft Palate Craniofac J 2000;37:447-452. Nasika MS. Epidemiological study of cleft lip/palate in the Greek population between 1980 and 1990. Doctoral thesis, Athens, 1997. Nyvad B, Fejerskov O. Baelum V. Visual-tactile caries diagnoses. In Dental Caries. Eds Fejerskov O. and Kidd Ed. (2nd Edition); Blackwell 2008:49-68. Silness J, Loe H. Periodontal disease in pregnancy. II. Correlation between oral hygiene and periodontal condition. Acta Odontol Scand 1964;22:112-135. Slayton RL, Williams L, Murray JC, et al. Genetic association studies of cleft lip and/or palate with hypodontia outside the cleft region. Cleft PalateCraniofacial 2003; 40: 274-279. Vanderas A. Inidence of cleft lip, cleft palate, and cleft lip and palate among races: a review. Cleft Palate J 1987;24:216-225. Vichi M, Franchi L. Abnormalities of the maxillary incisors in children with cleft lip and palate. ASDC J Dent Child 1995;62:412-417. 89 European Archives of Paediatric Dentistry // 10 (2). 2009