Polymicrogyria and myoclonic epilepsy in autosomal recessive cutis laxa type 2A

Neurogenetics. 2016 Oct;17(4):251-257. doi: 10.1007/s10048-016-0491-3. Epub 2016 Sep 8.

Abstract

Cutis laxa syndromes are rare inherited disorders of skin and connective tissue metabolism associated with variable systemic involvement. The main clinical manifestation is loose, wrinkled, redundant, inelastic skin, hypotonia, typical facies including short nose and down-slanting palpebral fissures, and varying degrees of developmental delay. The aim of this report is to describe two siblings diagnosed with a moderate form of ATP6V0A2-related cutis laxa with polymicrogyria (cobblestone-like brain dysgenesis). One of the patients has myoclonic epilepsy which may have contributed to his more severe clinical presentation. The literature on cutis laxa syndromes is reviewed.

Keywords: Autosomal recessive cutis laxa type 2A; Myoclonic epilepsy; Polymicrogyria.

Publication types

  • Case Reports

MeSH terms

  • Brain / diagnostic imaging
  • Brain / pathology
  • Brain / physiopathology
  • Child
  • Cutis Laxa / complications
  • Cutis Laxa / diagnostic imaging
  • Cutis Laxa / pathology*
  • Cutis Laxa / physiopathology*
  • Epilepsies, Myoclonic / complications
  • Epilepsies, Myoclonic / diagnostic imaging
  • Epilepsies, Myoclonic / pathology*
  • Epilepsies, Myoclonic / physiopathology*
  • Female
  • Humans
  • Male
  • Mutation
  • Polymicrogyria / complications
  • Polymicrogyria / diagnostic imaging
  • Polymicrogyria / pathology*
  • Polymicrogyria / physiopathology*
  • Siblings

Supplementary concepts

  • Cutis Laxa, Autosomal Recessive, Type IIA