Capillary malformation-arteriovenous malformation syndrome associated with basilar artery aneurysm

Pediatr Dermatol. 2022 Jul;39(4):662-663. doi: 10.1111/pde.15001. Epub 2022 May 3.

Abstract

A 23-day-old boy with prenatal diagnosis of basilar artery aneurysm presented with multiple congenital red patches consistent with capillary malformations. Genetic testing confirmed the presence of a heterozygous pathogenic variant of the RASA1 gene, confirming the diagnosis of capillary malformation-arteriovenous malformation (CM-AVM) syndrome. This case illustrates an atypical presentation of the RASA1 associated CM-AVM syndrome, with the intracranial vascular malformation diagnosis preceding the identification of the skin lesions. Arterial aneurysms have been associated with CM-AVM syndrome in rare instances but to our knowledge this is the first reported case of an aneurysm of the basilar artery.

Keywords: RASA1; arteriovenous malformation; capillary malformation; mutation; vascular malformations.

Publication types

  • Case Reports

MeSH terms

  • Arteriovenous Malformations
  • Capillaries / abnormalities
  • Humans
  • Intracranial Aneurysm* / complications
  • Intracranial Aneurysm* / diagnosis
  • Intracranial Aneurysm* / genetics
  • Male
  • Mutation
  • Port-Wine Stain* / complications
  • Port-Wine Stain* / diagnosis
  • Port-Wine Stain* / genetics
  • p120 GTPase Activating Protein / genetics

Substances

  • RASA1 protein, human
  • p120 GTPase Activating Protein

Supplementary concepts

  • Capillary Malformation-Arteriovenous Malformation