Novel DCX mutation-caused lissencephaly in a boy and very mild heterotopia in his mother

Pediatr Int. 2015 Apr;57(2):321-3. doi: 10.1111/ped.12502.

Abstract

We describe a novel mutation in DCX in a family in which a proband boy had classical lissencephaly and his mother had extremely mild subcortical band heterotopia. No factors that would make the mother's symptoms milder, such as somatic mosaicism or skewed X chromosome inactivation, were observed. From this family, we conclude that a DCX mutation causes a pleiotropic phenotype in the female even if X chromosome inactivation pattern is not skewed, and the novel missense mutation in DCX produced relatively mild dysfunction of the doublecortin protein.

Keywords: DCX; X chromosome inactivation; doublecortin; lissencephaly; subcortical band heterotopia.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Brain / pathology*
  • Child, Preschool
  • Classical Lissencephalies and Subcortical Band Heterotopias / genetics*
  • Doublecortin Domain Proteins
  • Doublecortin Protein
  • Female
  • Genetic Testing
  • Humans
  • Magnetic Resonance Imaging
  • Male
  • Microtubule-Associated Proteins / genetics*
  • Mothers
  • Mutation*
  • Neuropeptides / genetics*
  • Phenotype

Substances

  • DCX protein, human
  • Doublecortin Domain Proteins
  • Doublecortin Protein
  • Microtubule-Associated Proteins
  • Neuropeptides