Unusual ribbon-like periventricular heterotopia with congenital cataracts in a Japanese girl

Am J Med Genet A. 2012 Mar;158A(3):674-7. doi: 10.1002/ajmg.a.34258. Epub 2012 Feb 7.

Abstract

Periventricular heterotopia (PH), clumps of neurons mislocated beside the ventricle, is caused by failure to initiate migration during embryogenesis. We report on a 32-month-old Japanese girl with a unique subtype of PH, namely ribbon-like PH. The patient presented with severe psychomotor developmental delay, intractable epilepsy, and congenital cataracts and developed West syndrome phenotype. Brain magnetic resonance imaging revealed a unique undulating form of PH, categorized as ribbon-like PH, and other brain malformations including simplified gyri and dysgenesis of the corpus callosum. There was no evidence of prenatal TORCH infection or associated syndrome. Array-based comparative genomic hybridization revealed no chromosomal rearrangements. Genetic analyses of the FLNA, DCX, ARX, LIS1, and TUBA1A genes showed no mutations. Although little is known about ribbon-like PH, the clinical manifestations in our patient clearly differed from those in other reported patients.

Publication types

  • Case Reports

MeSH terms

  • Cataract / complications
  • Cataract / congenital*
  • Cataract / genetics
  • Child, Preschool
  • Comparative Genomic Hybridization
  • Female
  • Humans
  • Japan
  • Mutation
  • Periventricular Nodular Heterotopia / complications*
  • Periventricular Nodular Heterotopia / genetics
  • Real-Time Polymerase Chain Reaction

Supplementary concepts

  • Periventricular Laminar Heterotopia