Clinical, histomorphological and therapeutic features of the Van der Woude Syndrome: literature review and presentation of an unusual case

Eur J Paediatr Dent. 2018 Mar;19(1):70-73. doi: 10.23804/ejpd.2018.19.01.13.

Abstract

Background: Van der Woude syndrome (VWS), an autosomal dominant condition associated with lower lip pits and/or cleft palate, is caused by mutations in the interferon regulatory factor 6 gene (lRF6 gene). The genetic alterations identified to date that contribute to expression of the syndrome are chiefly mutations located on chromosome 1 (the largest of our chromosomes), mutations at p36 that codifies the gene GRHL (grainy-head transcriptor factor) and mutations involving IRF6 (interferon regulatory factor). With frequency ranging from 1:35,000 to 1:100,000, depending on ethnicity, gender, and socio-economic status, the syndrome accounts for about 2% of orofacial clefts. The clinical and histomorphological aspects of VWS are studied, and a case of heterozygous female twins of whom only one was affected with VWS is reported.

Conclusion: This very rare case (no similar case has been reported to date) contributes further evidence on modifying factors in the expression of this condition.

Publication types

  • Case Reports
  • Review

MeSH terms

  • Abnormalities, Multiple / diagnosis*
  • Abnormalities, Multiple / surgery*
  • Child, Preschool
  • Cleft Lip / diagnosis*
  • Cleft Lip / surgery*
  • Cleft Palate / diagnosis*
  • Cleft Palate / surgery*
  • Cysts / diagnosis*
  • Cysts / surgery*
  • Diagnosis, Differential
  • Female
  • Humans
  • Lip / abnormalities*
  • Lip / surgery*

Supplementary concepts

  • Van der Woude syndrome