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Tooth Agenesis Patterns in Bilateral Cleft Lip and Palate

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Tooth Agenesis Patterns in Bilateral Cleft Lip and Palate Eur J Oral Sci 2010; 118: 47–52 Ó 2010 The Authors. Printed in Singapore. All rights reserved Journal compilation Ó 2010 Eur J Oral Sci European Journal of Oral Sciences Theodosia N. Bartzela1, Carine E.L. Tooth agenesis patterns in bilateral Carels2, Ewald M. Bronkhorst3, Elisabeth Rønning4, Sara Rizell5, cleft lip and palate Anne Marie Kuijpers-Jagtman1,6 1Department of Orthodontics and Oral Biology, Radboud University Nijmegen Medical Centre, 2 Bartzela TN, Carels CEL, Bronkhorst EM, Rønning E, Rizell S, Kuijpers-Jagtman AM. Nijmegen, the Netherlands; Department of Orthodontics, Catholic University of Leuven, Tooth agenesis patterns in bilateral cleft lip and palate. Eur J Oral Sci 2010; 118: 47–52. Leuven, Belgium; 3Department of Cariology Ó 2010 The Authors. Journal compilation Ó 2010 Eur J Oral Sci and Preventive Dentistry, Radboud University Nijmegen, Medical Centre, Nijmegen, the Individuals with cleft lip and palate present significantly more dental anomalies, even Netherlands; 4Oslo Cleft Team, Department of Plastic Surgery, National Hospital, and Bredtvet outside the cleft area, than do individuals without clefts. Our aim was to evaluate the 5 prevalence of tooth agenesis and patterns of hypodontia in a large sample of patients Resource Centre, Oslo, Norway; Section of Jaw Orthopedics, University Clinics of with complete bilateral cleft lip and palate (BCLP). Serial panoramic radiographs (the Odontology, Gothenburg, Sweden; 6Cleft first radiograph was taken at 10.5–13.5 yr of age) of 240 patients with BCLP (172 male Palate Craniofacial Unit, Radboud University patients, 68 female patients) were examined. Third molars were not included in the Nijmegen Medical Centre, Nijmegen, the evaluation. Agenesis of at least one tooth was present in 59.8% of patients. Upper Netherlands laterals and upper and lower second premolars were missing most frequently. Using the tooth agenesis code (TAC), 52 different agenesis patterns were identified, of which Anne Marie Kuijpers-Jagtman, Department of simultaneous agenesis of 12, 22, 15, 25, 35, and 45 was the most frequent pattern. Nine Orthodontics and Oral Biology, Radboud of the 240 patients showed combined BCLP and oligodontia. University Nijmegen Medical Center, 309 Dentistry, PO Box 9101, 6500 HB Nijmegen, the Netherlands Telefax: +31–24–3540631 E-mail: [email protected] Key words: bilateral cleft lip and palate; cleft palate; dental agenesis; prevalence Accepted for publication October 2009 The prevalence of tooth agenesis (excluding third mo- dentition are small, ranging from 10 to 125, and often lars) in the general population differs according to con- involve other cleft types in addition to BCLP. The most tinent and gender, and ranges from 3.2 to 5.5% for men frequently missing single teeth in patients with BCLP are and from 4.6 to 7.6% for women (1). The mandibular the maxillary lateral incisors (9–11). Furthermore, few second premolar is the tooth most commonly affected studies have identified tooth agenesis patterns as op- (2.9–3.2%), followed by the maxillary lateral (1.6–1.8%), posed to reporting the prevalence of single missing teeth and the maxillary second premolar (1.4–1.6%). In (3, 11, 12), and only one study determined tooth agenesis patients with cleft lip and palate (CLP), as well as in their patterns for the entire dentition, but the sample size was non-affected siblings, tooth agenesis is more common small (12). Pattern recognition of tooth agenesis is than in the general population (2), which can be important because careful subphenotyping of CLP explained by the close relationship between tooth and patients based on dental development characteristics cleft formation with respect to developmental timing and might identify cleft subgroups, which could help to anatomical location. The maxillary lateral incisors are determine specific genetic contributions and identify the teeth most commonly missing in the cleft area, with a disease-causing alleles (5). prevalence ranging from 56.1 to 74% (3, 4). The preva- Therefore, the aim of this study was to investigate the lence of tooth agenesis outside the cleft region in cleft prevalence of agenesis for each tooth type and to identify patients is 27% (2, 5), with the second premolars being hypodontia patterns in patients with complete BCLP. affected most often (5, 6). The frequency of dental anomalies seems to be linked to the severity of the cleft malformation (2, 7). In the Material and methods most severe type of non-syndromic clefts, complete bilateral cleft lip and palate (BCLP) , the prevalence of Participants missing teeth reaches 100% (8). However, little data are The study comprised radiographs of 240 patients with available for this type of cleft. Table S1 provides an complete BCLP from the Cleft Palate Centers in Gothen- overview of the existing studies on BCLP. In general, burg (Sweden), Nijmegen (the Netherlands), and Oslo sample sizes of studies concerning the permanent (Norway). Inclusion criteria were as follows: 48 Bartzela et al. Table 1 Schematic representation of the human dentition and application of the binary system used to assign unique values to the pattern of tooth agenesis (13) Maxilla right (Q1) Maxilla left (Q2) A 18* 17 16 15 14 13 12 11 21 22 23 24 25 26 27 28* B 128 64 32 16 8 4 2 1 1 2 4 8 16 32 64 128 A 48* 47 46 45 44 43 42 41 31 32 33 34 35 36 37 38* Mandible right (Q4) Mandible left (Q3) Line A: tooth numbering according to the Fe´de´ration Dentaire Internationale (FDI) tooth numbering system, for maxilla (upper) and mandible (lower). Line B: values associated with missing teeth. Q1, Q2, Q3, Q4: first to fourth quadrants of the dentition. *Not included in this study. (i) Non-syndromic complete BCLP; diagnosis confirmed by the pre-operative record or by neonatal pictures Statistical analysis and/or study models. Patients with Simonart‘s band(s) Intra-observer and interobserver agreements were calcu- were included only if no hard tissue union was present. lated using kappa statistics (Table 2 describes the reliability (ii) No other associated congenital malformations, or statistics). Tooth counts and percentages were used to mental retardation. characterize tooth agenesis. The chi-square test (FisherÕs (iii) Caucasian ethnic background. exact test) was used to evaluate the relationship between (iv) At least 11 yr of age when the data were collected. The the prevalence of agenesis and other dichotomous vari- data collection for the three Cleft Palate Centers had ables, such as gender, left/right quadrant, and upper/lower covered the following time span: Oslo 1969–2008; jaw. Nijmegen 1970–2008; and Gothenburg 1980–2008. Methods Results Radiographs, taken according to the routine clinical pro- Prevalence cedure of each CLP center, were selected from the patient files. Each radiograph was allocated a random identification Records from 240 patients with BCLP (68 female number. No additional radiographs were taken. At least patients and 172 male patients) were evaluated. three panoramic radiographs [orthopantomograms According to data provided by the centers, a number of (OPTs)], taken at different ages of the patient, were avail- patients were excluded because of ethnicity (six patients able for each patient. Congenitally missing teeth were from Gothenburg cleft center, four patients from identified on OPTs and were verified by dental records to exclude premature extractions. Third molars were not included in the assessment. All radiographs were scored by Table 2 one observer (TB). Sixty radiographs were scored twice to assess intra-observer reliability. For assessing interobserver Kappa values for intra-observer and interobserver agreement for reliability, the same 60 radiographs were scored by three each tooth in the upper and lower arches; corresponding teeth in observers. the right and left quadrants were combined The method developed by van Wijk & Tan (13) was used to describe patterns of tooth agenesis. A binary system was Corresponding Intra-observer Interobserver teeth* agreement agreement applied to establish unique numbers associated with differ- ent patterns of tooth agenesis; the scoring was dichotomized Maxilla as presence (0) or absence (1) of teeth. A specific value was 7 0.00 1.00 assigned to each missing tooth type. The values were sum- 6 1.00 0.00 med for each quadrant, giving a unique value for each 5 0.87 0.72 pattern of hypodontia, the so-called tooth agenesis code 4 0.66 0.65 (TAC). According to the TAC, a certain quadrant without 3 1.00 0.67 tooth agenesis would have a TAC value of 0 and a quadrant 2 0.80 0.64 with complete tooth agenesis would have a TAC value of 1 0.90 0.28 255 (Table 1) shows the TAC coding system. The overall Mandible TAC score was used to identify patterns of tooth agenesis 7 0.00 0.00 for the entire mouth. For example, when the TAC = 6 0.00 0.00 100.123.038.001, the number 100 corresponds to the first 5 0.92 0.96 quadrant, 123 corresponds to the second quadrant, 038 4 0.00 0.00 corresponds to the third quadrant, and 001 corresponds to 3 0.00 0.00 the fourth quadrant (14). 2 0.66 0.39 1 0.49 )0.10 This research was been conducted in full accordance with ethical principles, including the World Medical Association *Teeth were numbered according to the Fe´de´ration Dentaire Declaration of Helsinki. Internationale (FDI) system. Tooth agenesis in BCLP 49 Table 3 Table 4 Tooth agenesis code (TAC), frequency and percentage of TAC, Thirty unique tooth agenesis code (TAC) patterns (frequency 1, corresponding missing teeth, and number (n) of missing teeth in
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