Retinitis pigmentosa associated with a dominant mutation in codon 46 of the peripherin/RDS gene (arginine-46-stop)

Am J Ophthalmol. 1995 Jan;119(1):65-71. doi: 10.1016/s0002-9394(14)73815-2.

Abstract

Purpose: We identified genetic mutations and characterized their associated phenotypes in patients with retinitis pigmentosa.

Methods: Patients with retinitis pigmentosa were prospectively examined and screened for genetic mutations.

Results: A 46-year-old man with retinitis pigmentosa was found to have a heterozygous mutation in the peripherin/RDS gene (arginine-46-stop). He had late onset of symptoms and demarcated peripheral retinal atrophy. All five first-degree relatives including his parents had no detectable mutations or retinitis pigmentosa. Genotypic data were consistent with reported family structure.

Conclusions: This study shows that new dominant mutations are a rare cause of isolated, or simplex, cases of retinitis pigmentosa. Identification of these mutations is helpful for genetic counseling.

Publication types

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

MeSH terms

  • Adult
  • Amino Acid Sequence
  • Arginine
  • Base Sequence
  • Codon / genetics*
  • DNA / analysis
  • Electroretinography
  • Fundus Oculi
  • Genes, Dominant*
  • Humans
  • Intermediate Filament Proteins / genetics*
  • Male
  • Membrane Glycoproteins*
  • Middle Aged
  • Molecular Sequence Data
  • Mutation*
  • Nerve Tissue Proteins*
  • Neuropeptides / genetics
  • Pedigree
  • Peripherins
  • Polymerase Chain Reaction
  • Prospective Studies
  • Retinal Degeneration / genetics*
  • Retinal Degeneration / pathology
  • Retinitis Pigmentosa / genetics*
  • Retinitis Pigmentosa / pathology
  • Rhodopsin / genetics

Substances

  • Codon
  • Intermediate Filament Proteins
  • Membrane Glycoproteins
  • Nerve Tissue Proteins
  • Neuropeptides
  • PRPH protein, human
  • PRPH2 protein, human
  • Peripherins
  • DNA
  • Rhodopsin
  • Arginine