Pyridoxine-dependent epilepsy in Tunisia is caused by a founder missense mutation of the ALDH7A1 gene

Gene. 2013 Apr 15;518(2):242-5. doi: 10.1016/j.gene.2013.01.041. Epub 2013 Jan 30.

Abstract

Pyridoxine-dependent epilepsy (PDE) is a rare autosomal recessive disorder characterized by seizures and therapeutic response to pharmacological dose of pyridoxine. Mutations in the ALDH7A1 gene, encoding α-aminoadipic semialdehyde (α-AASA) dehydrogenase (antiquitin), have been reported to cause PDE in most patients. In this study molecular analysis of seven PDE Tunisian patients revealed a common missense c.1364T>C mutation in the ALDH7A1 gene. The identification of a cluster of PDE pedigrees carrying the c.1364T>C mutation in a specific area raises the question of the origin of this mutation from a common ancestor. We carried out a genotype-based analysis by way of genotyping a new generated microsatellite marker within the ALDH7A1 gene. Genotype reconstruction of all affected pedigree members indicate that all c.1364T>C mutation carriers harbored the same allele, indicating a common ancestor. The finding of a founder effect in a rare disease is essential for the genetic diagnosis and the genetic counseling of affected PDE pedigrees in Tunisia.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aldehyde Dehydrogenase / genetics*
  • Epilepsy / genetics*
  • Founder Effect
  • Genotype
  • Humans
  • Male
  • Microsatellite Repeats*
  • Mutation, Missense*
  • Pyridoxine / therapeutic use
  • Tunisia

Substances

  • ALDH7A1 protein, human
  • Aldehyde Dehydrogenase
  • Pyridoxine

Supplementary concepts

  • Pyridoxine-dependent epilepsy