Angiodysplasia with osteohypertrophy affecting the oromaxillofacial area: clinical findings

J Craniofac Surg. 2001 Sep;12(5):485-9. doi: 10.1097/00001665-200109000-00016.


The authors report a case of oro-facial port-wine stain angiomatosis with cutaneous and mucosal lesions localized in the first and the second branch surface distribution areas of the right trigeminal nerve in association with right upper and lower lip, cheek, and hemimaxillary true hypertrophy leading to a severe dento-skeletal malocclusion with openbite and facial asymmetry. The authors through a revision of the Bibliography locate nosologically this case among the rare congenital angiodysplastic syndromes affecting the maxillo-facial district as the Sturge-Weber and the Klippel-Trenaunay syndromes. They debate also about etiopathogenetic, clinical, differential diagnosis, and therapeutic aspects concerning the treatment of patients affected by this kind of malformation.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Humans
  • Macroglossia / etiology
  • Magnetic Resonance Imaging
  • Male
  • Malocclusion / etiology
  • Sturge-Weber Syndrome / complications
  • Sturge-Weber Syndrome / pathology*