title: Three-dimensional analysis of tooth dimensions in the MSX1-missense mutation.
abstract: OBJECTIVE
  A novel, 3D technique to measure the differences in tooth crown morphology between the MSX1 cases and non-affected controls was designed to get a better understanding of dental phenotype-genotype associations.
  
  
  METHODS
  Eight Dutch subjects from a single family with tooth agenesis, all with an established nonsense mutation c.332 C > A, p. Ser 111 Stop in exon 1 of MSX1, were compared with unaffected controls regarding several aspects of tooth crown morphology of incisor and molar teeth. A novel method of quantitative three-dimensional analysis was used to detect differences.
  
  
  RESULTS
  Statistically significant shape differences were observed for the maxillary incisor in the MSX1 family compared with the controls on the following parameters: surface area, buccolingual dimension, squareness, and crown volume (P ≤ 0.002). Molar crown shape was unaffected.
  
  
  CONCLUSIONS
  A better understanding of dental phenotype-genotype associations may contribute to earlier diagnosis of some multiple-anomaly congenital syndromes involving dental anomalies.
  
  
  CONCLUSIONS
  A "shape database" that includes associated gene mutations resulting from developmental syndromes may facilitate the genetic identification of hypodontia cases.
authors.affiliation: Department of Oral and Maxillofacial Surgery, Prosthodontics and Special Dental Care, University Medical Centre Utrecht, Utrecht, The Netherlands. mcreton@umcutrecht.nl
authors.forename: Marijn
authors.initials: M
authors.lastname: Créton
authors.forename: Marie-José
authors.initials: MJ
authors.lastname: van den Boogaard
authors.forename: Thomas
authors.initials: T
authors.lastname: Maal
authors.forename: Luc
authors.initials: L
authors.lastname: Verhamme
authors.forename: Willem
authors.initials: W
authors.lastname: Fennis
authors.forename: Carine
authors.initials: C
authors.lastname: Carels
authors.forename: Anne Marie
authors.initials: AM
authors.lastname: Kuijpers-Jagtman
authors.forename: Marco
authors.initials: M
authors.lastname: Cune
mesh_terms: D000293:Adolescent; D000328:Adult; D000368:Aged; D000704:Analysis of Variance; D000848:Anodontia / Q000235:genetics*; D016022:Case-Control Studies; D002648:Child; D005260:Female; D006801:Humans; D021621:Imaging, Three-Dimensional / Q000379:methods*; D007180:Incisor; D051957:MSX1 Transcription Factor / Q000235:genetics*; D008297:Male; D003765:Models, Dental; D008963:Molar; D020125:Mutation, Missense; D009426:Netherlands; D009811:Odontometry / Q000379:methods; D018709:Statistics, Nonparametric; D014071:Tooth Abnormalities / Q000235:genetics / Q000473:pathology; D019228:Tooth Crown / Q000473:pathology*; D055815:Young Adult
chemical_list: D051957:MSX1 Transcription Factor