C9-R95X polymorphism in patients with neovascular age-related macular degeneration

Invest Ophthalmol Vis Sci. 2012 Jan 31;53(1):508-12. doi: 10.1167/iovs.11-8425.

Abstract

Purpose: A non-sense mutation at codon 95 in the gene encoding complement factor C9 (C9-R95X) is found most frequently among Japanese. The authors investigated the association between C9-R95X and Japanese patients with neovascular age-related macular degeneration (AMD) and polypoidal choroidal vasculopathy (PCV).

Methods: The presence of the C9-R95X polymorphism was assessed by direct sequencing in Japanese patients with either PCV (n = 105) or neovascular AMD (n = 198) and 396 control subjects. Multivariate regression analyses were conducted. Photocoagulation was applied in the eyes of mice with a heterozygous defect in the C3 gene and control wild-type mice. Photocoagulation was also applied to wild-type mice before either anti-C9 antibody or isotype IgG was injected into the eyes. The eyes were collected later for measurement of vascular endothelial growth factor (VEGF) and histological evaluation of choroidal neovascularization (CNV).

Results: The frequency of those with one or two C9-R95X variants was lower in neovascular AMD (2.02%) than in PCV (5.71%) and controls (6.05%). The presence of C9-R95X conferred a 4.7-fold reduction (95% confidence interval, 1.2-18.1; P = 0.021) in the risk for neovascular AMD after adjusting for the major AMD risk factors. A heterozygous defect in the C3 gene was associated with the reduced growth of laser-induced CNV, as was intraocular injection of anti-C9 antibody. This reduced CNV growth was accompanied by a decreased level of secreted VEGF in the intraocular fluid.

Conclusions: These findings support the notion that the haploinsufficiency of C9, a terminal complement complex component, engenders reduced intraocular secretion of VEGF and decreased risk for CNV development.

Publication types

  • Comparative Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Aged
  • Animals
  • Complement C9 / genetics*
  • Complement C9 / metabolism
  • DNA / genetics*
  • Disease Models, Animal
  • Female
  • Fluorescein Angiography
  • Fundus Oculi
  • Genotype
  • Humans
  • Macular Degeneration / genetics*
  • Macular Degeneration / metabolism
  • Macular Degeneration / pathology
  • Male
  • Mice
  • Mice, Inbred C57BL
  • Polymerase Chain Reaction
  • Polymorphism, Genetic*
  • Retinal Neovascularization / genetics*
  • Retinal Neovascularization / metabolism
  • Retinal Neovascularization / pathology

Substances

  • Complement C9
  • DNA