Novel RDH12 sequence variations in Leber congenital amaurosis

J AAPOS. 2010 Aug;14(4):349-51. doi: 10.1016/j.jaapos.2010.04.010.

Abstract

Leber congenital amaurosis (LCA) designates a severe congenital retinal dystrophy generally inherited in an autosomal-recessive manner and accounting for 5% of inherited retinopathies. Its main clinical features are severe visual loss, sensory nystagmus, amaurotic pupils, and unrecordable electroretinographic response. LCA has been associated with sequence variations of 14 different genes; in approximately 30% of all cases pathogenic mutations remain to be determined. We report 2 patients with a clinical phenotype of LCA associated with novel mutations of the RDH12 gene.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Alcohol Oxidoreductases / genetics*
  • Alcohol Oxidoreductases / metabolism
  • Child
  • DNA / genetics*
  • Diagnosis, Differential
  • Electroretinography
  • Female
  • Gene Frequency
  • Genetic Predisposition to Disease
  • Humans
  • Leber Congenital Amaurosis / diagnosis
  • Leber Congenital Amaurosis / genetics*
  • Male
  • Mutation*
  • Phenotype
  • Polymerase Chain Reaction

Substances

  • DNA
  • Alcohol Oxidoreductases
  • RDH12 protein, human