Moyamoya angiopathy in PHACE syndrome not associated with RNF213 variants

Childs Nerv Syst. 2019 Jul;35(7):1231-1237. doi: 10.1007/s00381-019-04145-9. Epub 2019 Apr 29.

Abstract

Moyamoya angiopathy is a rare vasculopathy with stenosis and/or occlusion of bilateral intracranial parts of internal carotid arteries and/or proximal parts of middle and anterior cerebral arteries. PHACE syndrome is characterized by large segmental hemangiomas in the cervical-facial region. Both conditions are known to be associated in rare cases. Recently, it was discussed in the literature that RNF213 variants could be etiologically involved in this association. Here, we describe a childhood case with this rare co-occurrence in which we did not identify any rare RNF213 variant. The clinical and genetic backgrounds are discussed.

Keywords: Moyamoya angiopathy; PHACE; Rare RNF 213 variants.

Publication types

  • Case Reports

MeSH terms

  • Adenosine Triphosphatases / genetics*
  • Adult
  • Aortic Coarctation / complications*
  • Aortic Coarctation / diagnostic imaging
  • Aortic Coarctation / genetics
  • Brain / diagnostic imaging
  • Eye Abnormalities / complications*
  • Eye Abnormalities / diagnostic imaging
  • Eye Abnormalities / genetics
  • Female
  • Humans
  • Magnetic Resonance Imaging
  • Moyamoya Disease / complications*
  • Moyamoya Disease / diagnostic imaging
  • Moyamoya Disease / genetics
  • Neurocutaneous Syndromes / complications*
  • Neurocutaneous Syndromes / diagnostic imaging
  • Neurocutaneous Syndromes / genetics
  • Ubiquitin-Protein Ligases / genetics*

Substances

  • RNF213 protein, human
  • Ubiquitin-Protein Ligases
  • Adenosine Triphosphatases

Supplementary concepts

  • PHACE association