Color-deficient cone mosaics associated with Xq28 opsin mutations: a stop codon versus gene deletions

Vision Res. 2010 Nov 23;50(23):2396-402. doi: 10.1016/j.visres.2010.09.015. Epub 2010 Sep 17.

Abstract

Our understanding of the etiology of red-green color vision defects is evolving. While missense mutations within the long- (L-) and middle-wavelength sensitive (M-) photopigments and gross rearrangements within the L/M-opsin gene array are commonly associated with red-green defects, recent work using adaptive optics retinal imaging has shown that different genotypes can have distinct consequences for the cone mosaic. Here we examined the cone mosaic in red-green color deficient individuals with multiple X-chromosome opsin genes that encode L opsin, as well as individuals with a single X-chromosome opsin gene that encodes L opsin and a single patient with a novel premature termination codon in his M-opsin gene and a normal L-opsin gene. We observed no difference in cone density between normal trichomats and multiple or single-gene deutans. In addition, we demonstrate different phenotypic effects of a nonsense mutation versus the previously described deleterious polymorphism, (LIAVA), both of which differ from multiple and single-gene deutans. Our results help refine the relationship between opsin genotype and cone photoreceptor mosaic phenotype.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Adult
  • Child
  • Codon, Terminator*
  • Color Vision Defects / genetics*
  • Color Vision Defects / physiopathology*
  • Cone Opsins / genetics*
  • Female
  • Gene Deletion*
  • Genes, X-Linked / genetics*
  • Genotype
  • Humans
  • Male
  • Middle Aged
  • Phenotype
  • Retina / pathology
  • Retinal Cone Photoreceptor Cells / pathology*
  • Sequence Analysis, DNA
  • Young Adult

Substances

  • Codon, Terminator
  • Cone Opsins