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Clin Genet ; 94(3-4): 330-338, 2018 10.
Artigo em Inglês | MEDLINE | ID: mdl-29888403

RESUMO

Genetic alterations in the complement system have been linked to a variety of diseases, including atypical hemolytic uremic syndrome (aHUS), C3 glomerulopathy (C3G), and age-related macular degeneration (AMD). We performed sequence analysis of the complement genes complement factor H (CFH), complement factor I (CFI), and complement C3 (C3) in 866 aHUS/C3G and 697 AMD patients. In total, we identified 505 low-frequency alleles, representing 121 unique variants, of which 51 are novel. CFH contained the largest number of unique low-frequency variants (n = 64; 53%), followed by C3 (n = 32; 26%) and CFI (n = 25; 21%). A substantial number of variants were found in both patients groups (n = 48; 40%), while 41 (34%) variants were found only in aHUS/C3G and 32 (26%) variants were AMD specific. Genotype-phenotype correlations between the disease groups identified a higher frequency of protein altering alleles in short consensus repeat 20 (SCR20) of factor H (FH), and in the serine protease domain of factor I (FI) in aHUS/C3G patients. In AMD, a higher frequency of protein-altering alleles was observed in SCR3, SCR5, and SCR7 of FH, the SRCR domain of FI, and in the MG3 domain of C3. In conclusion, we observed a substantial overlap of variants between aHUS/C3G and AMD; however, there is a distinct clustering of variants within specific domains.


Assuntos
Síndrome Hemolítico-Urêmica Atípica/genética , Complemento C3/genética , Fator I do Complemento/genética , Genótipo , Glomerulonefrite Membranosa/genética , Degeneração Macular/genética , Fenótipo , Síndrome Hemolítico-Urêmica Atípica/metabolismo , Síndrome Hemolítico-Urêmica Atípica/fisiopatologia , Estudos de Coortes , Complemento C3/metabolismo , Fator H do Complemento/genética , Fator H do Complemento/metabolismo , Fator I do Complemento/metabolismo , Predisposição Genética para Doença , Glomerulonefrite Membranosa/metabolismo , Glomerulonefrite Membranosa/fisiopatologia , Humanos , Degeneração Macular/metabolismo , Degeneração Macular/fisiopatologia
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