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Molecular Immunology
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Genetic deficiency of complement factor H in a patient with age-related macular degeneration and membranoproliferative glomerulonephritis

Authors: Montes, Tamara; Goicoechea de Jorge, Elena; Ramos, Rosa; Gomá, Montserrat; Pujol, Octavi; Sánchez-Corral, Pilar; Rodríguez de Córdoba, Santiago;

Genetic deficiency of complement factor H in a patient with age-related macular degeneration and membranoproliferative glomerulonephritis

Abstract

Age-related macular degeneration (AMD) and membranoproliferative glomerulonephritis type II (MPGN2) are dense deposit diseases that share a genetic association with complement genes and have complement proteins as important components of the dense deposits. Here, we present the case of a 64-year-old smoker male who developed both AMD and MPGN2 in his late 50s. The patient presented persistent low plasma levels of C3, factor H levels in the lower part of the normal range and C3NeF traces. Genetic analyses of the CFH, CFB, C3, CFHR1-CFHR3 and LOC387715/HTRA1 genes revealed that the patient was heterozygote for a novel missense mutation in exon 9 of CFH (c.1292 G>A) that results in a Cys431Tyr substitution in SCR7 of the factor H protein. In addition, he was homozygote for the His402 CFH allele, heterozygote for the Ser69 LOC387715 allele, homozygote for the Arg32 (BFS) CFB allele, heterozygote for the Gly102 (C3F) C3 allele and carried no deletion of the CFHR1/CFHR3 genes. Proteomic and functional analyses indicate absence in plasma of the factor H allele carrying the Cys431Tyr mutation. As a whole, these data recapitulate a prototypical complement genetic profile, including a partial factor H deficiency and the presence of major risk factors for AMD and MPGN2, which support the hypothesis that these dense deposit diseases have a common pathogenic mechanism involving dysregulation of the alternative pathway of complement activation.

Country
Spain
Keywords

Glomerulonephritis, Membranoproliferative, Blotting, Western, Molecular Sequence Data, Complement, Eye, Kidney, Mass Spectrometry, Macular Degeneration, Alternative pathway, Chlorocebus aethiops, Animals, Humans, Genetic Predisposition to Disease, Cysteine, Membranoproliferative glomerulonephritis, Base Sequence, Age-related macular degeneration, Factor H, Middle Aged, Recombinant Proteins, Human mutation, Amino Acid Substitution, Complement Factor H, COS Cells, Mutation, Tyrosine

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citations
This is an alternative to the "Influence" indicator, which also reflects the overall/total impact of an article in the research community at large, based on the underlying citation network (diachronically).
BIP!Citations provided by BIP!
popularity
This indicator reflects the "current" impact/attention (the "hype") of an article in the research community at large, based on the underlying citation network.
BIP!Popularity provided by BIP!
influence
This indicator reflects the overall/total impact of an article in the research community at large, based on the underlying citation network (diachronically).
BIP!Influence provided by BIP!
impulse
This indicator reflects the initial momentum of an article directly after its publication, based on the underlying citation network.
BIP!Impulse provided by BIP!
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