Literature DB >> 27026170

Novel Mutations Causing C5 Deficiency in Three North-African Families.

Roger Colobran1,2, Clara Franco-Jarava3, Andrea Martín-Nalda4, Neus Baena5, Elisabeth Gabau5, Natàlia Padilla6, Xavier de la Cruz6, Ricardo Pujol-Borrell3, David Comas7, Pere Soler-Palacín4, Manuel Hernández-González3.   

Abstract

The complement system plays a central role in defense to encapsulated bacteria through opsonization and membrane attack complex (MAC) dependent lysis. The three activation pathways (classical, lectin, and alternative) converge in the cleavage of C5, which initiates MAC formation and target lysis. C5 deficiency is associated to recurrent infections by Neisseria spp. In the present study, complement deficiency was suspected in three families of North-African origin after one episode of invasive meningitis due to a non-groupable and two uncommon Meningococcal serotypes (E29, Y). Activity of alternative and classical pathways of complement were markedly reduced and the measurement of terminal complement components revealed total C5 absence. C5 gene analysis revealed two novel mutations as causative of the deficiency: Family A propositus carried a homozygous deletion of two adenines in the exon 21 of C5 gene, resulting in a frameshift and a truncated protein (c.2607_2608del/p.Ser870ProfsX3 mutation). Families B and C probands carried the same homozygous deletion of three consecutive nucleotides (CAA) in exon 9 of the C5 gene, leading to the deletion of asparagine 320 (c.960_962del/p.Asn320del mutation). Family studies confirmed an autosomal recessive inheritance pattern. Although sharing the same geographical origin, families B and C were unrelated. This prompted us to investigate this mutation prevalence in a cohort of 768 North-African healthy individuals. We identified one heterozygous carrier of the p.Asn320del mutation (allelic frequency = 0.065 %), indicating that this mutation is present at low frequency in North-African population.

Entities:  

Keywords:  African continental ancestry group; Complement system; complement 5 deficiency; meningococcal disease; mutation

Mesh:

Substances:

Year:  2016        PMID: 27026170     DOI: 10.1007/s10875-016-0275-4

Source DB:  PubMed          Journal:  J Clin Immunol        ISSN: 0271-9142            Impact factor:   8.317


  27 in total

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6.  Meningococcal meningitis in familial deficiency of the fifth component of complement.

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