Incomplete penetrance with normal MRI in a woman with germline mutation of the DCX gene

Neurology. 2001 Jul 24;57(2):327-30. doi: 10.1212/wnl.57.2.327.

Abstract

X-linked isolated lissencephaly sequence (ILS) and subcortical band heterotopia are allelic human disorders associated with mutations of the DCX gene in both familial and sporadic forms. The authors describe a large Sardinian family in which three brothers with ILS have a missense mutation of the DCX gene. Their mother, a nonmosaic carrier, has a normal phenotype and cranial MRI. Skewed X-inactivation in the lymphocytes was also ruled out. This is the first report of an asymptomatic carrier of a DCX mutation likely due to apparent nonpenetrance.

Publication types

  • Research Support, U.S. Gov't, P.H.S.

MeSH terms

  • Adolescent
  • Brain / pathology*
  • Brain Diseases / genetics*
  • Brain Diseases / pathology*
  • Child
  • Child, Preschool
  • Doublecortin Domain Proteins
  • Doublecortin Protein
  • Female
  • Genetic Linkage / genetics
  • Germ-Line Mutation / genetics*
  • Humans
  • Magnetic Resonance Imaging
  • Microtubule-Associated Proteins*
  • Neuropeptides / genetics*
  • Pedigree
  • Penetrance*
  • X Chromosome / genetics*

Substances

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