title: The association between WNT10A variants and dental development in patients with isolated oligodontia.
abstract: In this study we aimed to determine the effect of WNT10A variants on dental development in patients with oligodontia. Forty-three (25 boys and 18 girls) individuals were eligible for this study. Stage of development for each present tooth was assessed using the Demirjian method. In case no corresponding tooth was present, regression equations were applied for dental age to be calculated. The ratio between length of root and length of crown was ascertained for each present tooth in all quadrants. All patients were physically examined by a clinical geneticist and DNA analysis of the WNT10A gene was performed. Linear regression models were applied to analyze the association between WNT10A variants and dental age. The same analysis was applied to study the association between WNT10A variants and root elongation for each present tooth. One ordinal regression model was applied to analyze the association between WNT10A variants and development of present maxillary and mandibular teeth. Thirty-six (84%) patients were detected with WNT10A variants of which six patients displayed evident ectodermal features. Dental age was 1.50 (95% confidence interval (CI): -2.59, -0.42) to 1.96 (95% CI: -3.76, -0.17) years lower in patients with WNT10A variants compared with patients without variants. The development of maxillary canine, maxillary second molar and mandibular second molar was statistically significantly delayed in patients with WNT10A variants compared with patients without variants. The impact of WNT10A variants on dental development increases with presence of the nonsense c.(321C>A p.(C107*)) variant and the number of missing teeth.
authors.affiliation: Department of Oral & Maxillofacial Surgery, Special Dental Care and Orthodontics, Erasmus University Medical Centre, Rotterdam, The Netherlands.|Department of Epidemiology, The Generation R Study Group, Erasmus University Medical Centre, Rotterdam, The Netherlands.
authors.forename: Brunilda
authors.initials: B
authors.lastname: Dhamo
authors.affiliation: Department of Oral and Maxillofacial Surgery, Prosthodontics and Special Dental Care, University Medical Centre Utrecht, Utrecht, The Netherlands.
authors.forename: Willem
authors.initials: W
authors.lastname: Fennis
authors.affiliation: Department of Oral and Maxillofacial Surgery, Prosthodontics and Special Dental Care, University Medical Centre Utrecht, Utrecht, The Netherlands.
authors.forename: Marijn
authors.initials: M
authors.lastname: Créton
authors.affiliation: Department of Oral & Maxillofacial Surgery, Special Dental Care and Orthodontics, Erasmus University Medical Centre, Rotterdam, The Netherlands.|Department of Epidemiology, The Generation R Study Group, Erasmus University Medical Centre, Rotterdam, The Netherlands.
authors.forename: Strahinja
authors.initials: S
authors.lastname: Vucic
authors.affiliation: Department of Oral Function and Prosthodontics, UMC Groningen, Groningen, The Netherlands.|Centre for Special Dental Care, St Antonius Hospital, Nieuwegein, The Netherlands.
authors.forename: Marco
authors.initials: M
authors.lastname: Cune
authors.affiliation: Department of Medical Genetics, University Medical Center Utrecht, Utrecht, The Netherlands.
authors.forename: Hans Kristian
authors.initials: HK
authors.lastname: Ploos van Amstel
authors.affiliation: Department of Oral & Maxillofacial Surgery, Special Dental Care and Orthodontics, Erasmus University Medical Centre, Rotterdam, The Netherlands.|Department of Epidemiology, The Generation R Study Group, Erasmus University Medical Centre, Rotterdam, The Netherlands.
authors.forename: Eppo B
authors.initials: EB
authors.lastname: Wolvius
authors.affiliation: Department of Medical Genetics, University Medical Center Utrecht, Utrecht, The Netherlands.
authors.forename: Marie-José
authors.initials: MJ
authors.lastname: van den Boogaard
authors.affiliation: Department of Oral & Maxillofacial Surgery, Special Dental Care and Orthodontics, Erasmus University Medical Centre, Rotterdam, The Netherlands. e.ongkosuwito@erasmusmc.nl.|Department of Epidemiology, The Generation R Study Group, Erasmus University Medical Centre, Rotterdam, The Netherlands. e.ongkosuwito@erasmusmc.nl.
authors.forename: Edwin M
authors.initials: EM
authors.lastname: Ongkosuwito
mesh_terms: D000293:Adolescent; D000848:Anodontia / Q000000981:diagnostic imaging / Q000235:genetics* / Q000473:pathology; D002648:Child; D018389:Codon, Nonsense / Q000235:genetics; D005260:Female; D006801:Humans; D008297:Male; D009154:Mutation; D010641:Phenotype; D014070:Tooth / Q000000981:diagnostic imaging / Q000382:microbiology / Q000503:physiopathology*; D051153:Wnt Proteins / Q000235:genetics*
chemical_list: D018389:Codon, Nonsense; C484142:WNT10A protein, human; D051153:Wnt Proteins