Phenotype variations within a choroideremia family lacking the entire CHM gene

Ophthalmic Genet. 1995 Dec;16(4):143-50. doi: 10.3109/13816819509057855.

Abstract

A Swedish family with choroideremia and a deletion of the CHM gene has been studied with ophthalmological examination, full-field electroretinography, and DNA analysis in order to characterize the phenotype of the disease. Although all four patients studied had a complete deletion of the gene, they showed a considerable variability regarding the phenotype, including the electroretinogram tracings. Two of the affected males demonstrated a severe form of choroideremia with low or nondetectable ERG recordings, while the other two affected males showed a less severe phenotype with only a slight reduction of the ERG amplitudes. The variation of the clinical phenotype among family members carrying the same mutation indicates that the severity of choroideremia is not solely a function of the CHM gene.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Aged
  • Child
  • Choroideremia / genetics*
  • Choroideremia / pathology
  • Choroideremia / physiopathology
  • DNA Mutational Analysis
  • Electrophoresis, Polyacrylamide Gel
  • Electroretinography
  • Female
  • Fundus Oculi
  • Gene Deletion
  • Genes*
  • Genetic Variation / genetics*
  • Humans
  • Male
  • Middle Aged
  • Mutation
  • Pedigree
  • Phenotype
  • Retina / pathology
  • Retina / physiopathology