Case report of segmental odontomaxillary dysplasia with cutaneous manifestations

Eur J Paediatr Dent. 2020 Sep;21(3):235-237. doi: 10.23804/ejpd.2020.21.03.14.

Abstract

Background: Segmental odontomaxillary dysplasia is an uncommon nonhereditary growth disorder that affects the maxilla, gums and ipsilateral dentition. The disorder is diagnosed mainly based on dental (over-retention of primary teeth, dental agenesis and diastemas) and bone findings (bone sclerosis, irregular trabeculation of immature bone and reduced maxillary sinus). This paper provides a case report.

Case report: A 5-year-old child with skin manifestations including hypertrichosis, facial erythema and pigmented nevus was diagnosed with type II segmental odontomaxillary dysplasia based on clinical, radiographic and histopathological analysis.

Conclusion: The skin findings can help with the suspicion of segmental odontomaxillary dysplasia, although the definitive diagnosis is typically established by a paediatric dentist based on clinical and radiological findings.

Publication types

  • Case Reports

MeSH terms

  • Child, Preschool
  • Diastema*
  • Humans
  • Maxilla
  • Odontodysplasia*
  • Skin Diseases*
  • Tooth, Deciduous