PHACES syndrome and multi-regional odontodysplasia: a case report

J Clin Pediatr Dent. 2024 May;48(3):166-170. doi: 10.22514/jocpd.2024.070. Epub 2024 May 3.

Abstract

PHACES syndrome is an acronym for the syndromic presentation of Posterior fossa malformation, Hemangioma, Arterial anomalies, Coarctation of aorta/cardiac defects, Eye abnormalities and Sternal malformations. Infantile hemangiomas are the most common tumors of infancy. Regional odontodysplasia, commonly referred to as "ghost teeth", is a rare localized developmental malformation of enamel and dentin with varying levels of severity that results in unusual clinical and radiographic appearances of affected teeth. This report describes a rare case of a two-year-old Caucasian male diagnosed with PHACES syndrome also presenting with multi-regional odontodysplasia. Ten of twenty teeth were dysplastic. The patient was treated under general anesthesia in a hospital setting. All affected primary teeth were extracted due to sensitivity, abscess and extremely poor long-term prognosis. Moving forward, a long-term interdisciplinary approach will be necessary to address this child's dentition as it develops.

Keywords: Dental anomaly; Odontodysplasia; PHACES syndrome.

Publication types

  • Case Reports

MeSH terms

  • Aortic Coarctation* / complications
  • Aortic Coarctation* / diagnostic imaging
  • Child, Preschool
  • Cranial Fossa, Posterior / abnormalities
  • Cranial Fossa, Posterior / diagnostic imaging
  • Eye Abnormalities* / complications
  • Humans
  • Male
  • Neurocutaneous Syndromes* / complications
  • Odontodysplasia* / diagnostic imaging
  • Tooth Extraction

Supplementary concepts

  • PHACE association