Macular dystrophy with protan genotype and phenotype studied with cone type specific ERGs

Curr Eye Res. 2001 Mar;22(3):221-8. doi: 10.1076/ceyr.22.3.221.5514.

Abstract

Purpose: To determine the L- and M-cone driven ERG responses in a male patient with macular dystrophy and a protan phenotype.

Methods: We measured large field ERG thresholds to stimuli which modulated exclusively the L- or the M-cones or the two in various combinations (both in-phase and in counterphase). In none of the stimuli, the S-cones were modulated. Additionally, standard and multifocal ERGs were measured. Analysis of the L- and M-cone pigment genes was performed by means of PCR, RFLP analysis and DNA sequencing techniques.

Results: Macular dystrophy was revealed by the markedly abnormal multifocal ERGs in presence of near normal standard ERGs. The large field ERG responses were exclusively driven by the M-cones with enlarged thresholds when compared with otherwise normal protanopes. In addition, the M-cone driven ERG response phases were abnormal. Pigment gene analysis confirmed a protan genotype with the presence of a single 5'red/3'green hybrid pigment gene.

Conclusions: Our novel stimulus technique allows a reliable analysis of the separate cone pathways even in cases with macular dysfunction. The increased thresholds and the abnormal phase behavior of the M-cone driven ERGs reflect altered mechanisms of the retinal physiology in this patient. The data strongly suggest that the macular dystrophy and the protanopia have independent origins.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Color Vision Defects / genetics
  • Color Vision Defects / physiopathology*
  • DNA Primers
  • Electroretinography*
  • Genotype
  • Humans
  • Macular Degeneration / genetics
  • Macular Degeneration / physiopathology*
  • Male
  • Phenotype
  • Polymerase Chain Reaction
  • Polymorphism, Restriction Fragment Length
  • Retinal Cone Photoreceptor Cells / physiopathology*
  • Retinal Pigments / genetics*
  • Sequence Analysis, DNA

Substances

  • DNA Primers
  • Retinal Pigments