Human complement factor H Y402H polymorphism causes an age-related macular degeneration phenotype and lipoprotein dysregulation in mice

Proc Natl Acad Sci U S A. 2019 Feb 26;116(9):3703-3711. doi: 10.1073/pnas.1814014116. Epub 2019 Feb 11.

Abstract

One of the strongest susceptibility genes for age-related macular degeneration (AMD) is complement factor H (CFH); however, its impact on AMD pathobiology remains unresolved. Here, the effect of the principal AMD-risk-associated CFH variant (Y402H) on the development and progression of age-dependent AMD-like pathologies was determined in vivo. Transgenic mice expressing equal amounts of the full-length normal human CFH Y402 (CFH-Y/0) or the AMD-risk associated CFH H402 (CFH-H/H) variant on a Cfh-/- background were aged to 90 weeks and switched from normal diet (ND) to a high fat, cholesterol-enriched (HFC) diet for 8 weeks. The resulting phenotype was compared with age-matched controls maintained on ND. Remarkably, an AMD-like phenotype consisting of vision loss, increased retinal pigmented epithelium (RPE) stress, and increased basal laminar deposits was detected only in aged CFH-H/H mice following the HFC diet. These changes were not observed in aged CFH-Y/0 mice or in younger (36- to 40-week-old) CFH mice of both genotypes fed either diet. Biochemical analyses of aged CFH mice after HFC diet revealed genotype-dependent changes in plasma and eyecup lipoproteins, but not complement activation, which correlated with the AMD-like phenotype in old CFH-H/H mice. Specifically, apolipoproteins B48 and A1 are elevated in the RPE/choroid of the aged CFH-H/H mice compared with age-matched control CFH-Y/0 fed a HFC diet. Hence, we demonstrate a functional consequence of the Y402H polymorphism in vivo, which promotes AMD-like pathology development and affects lipoprotein levels in aged mice. These findings support targeting lipoproteins as a viable therapeutic strategy for treating AMD.

Keywords: RPE; age-related macular degeneration; complement; genetic risk; lipoproteins.

Publication types

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

MeSH terms

  • Animals
  • Complement Activation / genetics*
  • Complement Factor H / genetics*
  • Diet, High-Fat / adverse effects
  • Female
  • Genotype
  • Humans
  • Lipoproteins / genetics*
  • Lipoproteins / metabolism
  • Macular Degeneration / genetics*
  • Macular Degeneration / pathology
  • Male
  • Mice
  • Mice, Transgenic / genetics
  • Polymorphism, Single Nucleotide / genetics
  • Retinal Pigment Epithelium / metabolism
  • Retinal Pigment Epithelium / pathology

Substances

  • Lipoproteins
  • Complement Factor H