Epileptic Encephalopathy in Adams-Oliver Syndrome Associated to a New DOCK6 Mutation: A Peculiar Behavioral Phenotype

Neuropediatrics. 2018 Jun;49(3):217-221. doi: 10.1055/s-0038-1639372. Epub 2018 Apr 9.

Abstract

Adams-Oliver syndrome (AOS) is characterized by a combination of congenital scalp defects (aplasia cutis congenita) and terminal transverse limb malformations of variable severity. When neurological findings are present, patients are reported as AOS variants. We describe a child with compound heterozygosity of the DOCK6 gene, aplasia cutis, terminal transverse limb defects, cardiovascular impairment, intellectual disability, and brain malformations with intracranial calcifications. He suffers from a severe refractory epileptic encephalopathy characterized by polymorphic seizures with prolonged periods of electroencephalogram (EEG), continuous epileptiform activity related to clinical inactivity, and closure of eyes with an "ON-OFF" behavior.

Publication types

  • Case Reports

MeSH terms

  • Brain / physiopathology
  • Ectodermal Dysplasia / genetics*
  • Ectodermal Dysplasia / physiopathology
  • Epilepsy / genetics*
  • Epilepsy / physiopathology
  • Guanine Nucleotide Exchange Factors / genetics*
  • Humans
  • Infant
  • Limb Deformities, Congenital / genetics*
  • Limb Deformities, Congenital / physiopathology
  • Male
  • Mutation*
  • Phenotype
  • Scalp Dermatoses / congenital*
  • Scalp Dermatoses / genetics
  • Scalp Dermatoses / physiopathology

Substances

  • DOCK6 protein, human
  • Guanine Nucleotide Exchange Factors

Supplementary concepts

  • Adams Oliver syndrome