Atypical pyridoxine-dependent epilepsy due to a pseudoexon in ALDH7A1

Mol Genet Metab. 2012 Apr;105(4):684-6. doi: 10.1016/j.ymgme.2012.01.011. Epub 2012 Jan 20.

Abstract

We report two siblings with atypical pyridoxine-dependant epilepsy, modest elevation of biomarkers, in which the open reading frame and the splice sites of ALDH7A1 did not show any mutations. Subsequent genetic analysis revealed a deep homozygous intronic mutation in ALDH7A1 resulting in two types of transcripts: the major transcript containing a pseudoexon, and the minor transcript representing the authentic spliced transcript. In future, this mutation may be targeted with antisense-therapy aiming at exclusion of the pseudoexon.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Aldehyde Dehydrogenase / genetics*
  • Epilepsy / diagnosis
  • Epilepsy / genetics*
  • Female
  • Genetic Testing
  • Homozygote
  • Humans
  • Infant, Newborn
  • Introns / genetics*
  • Male
  • Mutation / genetics*
  • RNA Splicing / genetics*
  • Siblings

Substances

  • ALDH7A1 protein, human
  • Aldehyde Dehydrogenase

Supplementary concepts

  • Pyridoxine-dependent epilepsy