Literature DB >> 22288589

Complete deficiency of the sixth complement component (C6Q0), susceptibility to Neisseria meningitidis infections and analysis of the frequencies of C6Q0 gene defects in South Africans.

A Orren1, E P Owen, H E Henderson, L van der Merwe, F Leisegang, C Stassen, P C Potter.   

Abstract

Complete complement component 6 deficiency (C6Q0) is a co-dominant genetic disease presenting as increased susceptibility to invasive Neisseria meningitidis infections. Affected individuals have two affected alleles which can be homozygous or compound heterozygous for the particular gene defects they carry. This disorder has been diagnosed relatively frequently in Western Cape South Africans. Affected patients are prescribed penicillin prophylaxis. In 2004 we commenced a clinical follow-up study of 46 patients. Of these, 43 had family age-matched C6 sufficient controls. Participants were classified as either (i) well, or (ii) having a serious illness (SI) or died (D). An SI was a long-term illness that did not allow the performance of normal daily activities. Among 43 patients, 21 were well and 22 were SI/D, while among 43 matched controls, 35 were well and eight were SI/D. This difference is highly significant. Among all 46 C6Q0 patients, those who had had recurrent infection had significantly more SI/D than those who had suffered none or one infection. Thus, this work demonstrates the long-term serious outcome of repeated meningococcal disease (MD) episodes. We investigated the frequencies of four C6Q0 pathogenic mutations known to affect Cape patients (828delG, 1138delC, 821delA and 1879delG) in 2250 newborns. A total of 103 defective alleles (2·28%) and three affected C6Q0 individuals were detected. For all defects combined, 5·24 affected subjects (C6Q0) are expected among 10,000 individuals. What is still unknown is the number of C6Q0 individuals who suffer MD or other infectious diseases.
© 2011 The Authors. Clinical and Experimental Immunology © 2011 British Society for Immunology.

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Year:  2012        PMID: 22288589      PMCID: PMC3374278          DOI: 10.1111/j.1365-2249.2011.04525.x

Source DB:  PubMed          Journal:  Clin Exp Immunol        ISSN: 0009-9104            Impact factor:   4.330


  17 in total

1.  Mutation nomenclature extensions and suggestions to describe complex mutations: a discussion.

Authors:  J T den Dunnen; S E Antonarakis
Journal:  Hum Mutat       Date:  2000       Impact factor: 4.878

2.  Prophylaxis against Neisseria meningitidis infections and antibody responses in patients with deficiency of the sixth component of complement.

Authors:  P C Potter; C E Frasch; W J van der Sande; R C Cooper; Y Patel; A Orren
Journal:  J Infect Dis       Date:  1990-05       Impact factor: 5.226

3.  The 'singles' method for segregation analysis under incomplete ascertainment.

Authors:  A M Davie
Journal:  Ann Hum Genet       Date:  1979-05       Impact factor: 1.670

4.  Functionally active complement proteins C6 and C7 detected in C6- and C7-deficient individuals.

Authors:  R Würzner; A Orren; P Potter; B P Morgan; D Ponard; P Späth; M Brai; M Schulze; L Happe; O Götze
Journal:  Clin Exp Immunol       Date:  1991-03       Impact factor: 4.330

Review 5.  Infections of people with complement deficiencies and patients who have undergone splenectomy.

Authors:  Sanjay Ram; Lisa A Lewis; Peter A Rice
Journal:  Clin Microbiol Rev       Date:  2010-10       Impact factor: 26.132

Review 6.  Complement deficiency states and infection: epidemiology, pathogenesis and consequences of neisserial and other infections in an immune deficiency.

Authors:  S C Ross; P Densen
Journal:  Medicine (Baltimore)       Date:  1984-09       Impact factor: 1.889

Review 7.  Infectious diseases associated with complement deficiencies.

Authors:  J E Figueroa; P Densen
Journal:  Clin Microbiol Rev       Date:  1991-07       Impact factor: 26.132

8.  Deficiency of the sixth component of complement and susceptibility to Neisseria meningitidis infections: studies in 10 families and five isolated cases.

Authors:  A Orren; P C Potter; R C Cooper; E du Toit
Journal:  Immunology       Date:  1987-10       Impact factor: 7.397

9.  Properties of a low molecular weight complement component C6 found in human subjects with subtotal C6 deficiency.

Authors:  A Orren; R Würzner; P C Potter; B A Fernie; S Coetzee; B P Morgan; P J Lachmann
Journal:  Immunology       Date:  1992-01       Impact factor: 7.397

10.  Antibodies to meningococcal class 1 outer membrane proteins in South African complement-deficient and complement-sufficient subjects.

Authors:  A Orren; R E Warren; P C Potter; A M Jones; P J Lachmann; J T Poolman
Journal:  Infect Immun       Date:  1992-11       Impact factor: 3.441

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  9 in total

Review 1.  Role of complement and potential of complement inhibitors in myasthenia gravis and neuromyelitis optica spectrum disorders: a brief review.

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Journal:  J Neurol       Date:  2019-09-03       Impact factor: 4.849

2.  Complement factor 5 (C5) p.A252T mutation is prevalent in, but not restricted to, sub-Saharan Africa: implications for the susceptibility to meningococcal disease.

Authors:  C Franco-Jarava; D Comas; A Orren; M Hernández-González; R Colobran
Journal:  Clin Exp Immunol       Date:  2017-04-20       Impact factor: 4.330

3.  Identification of a novel mutation in the C6 gene of a Han Chinese C6SD child with meningococcal disease.

Authors:  Ai-Qian Zhang; Yu-Xing Liu; Jie-Yuan Jin; Chen-Yu Wang; Liang-Liang Fan; Da-Bao Xu
Journal:  Exp Ther Med       Date:  2021-03-19       Impact factor: 2.447

4.  Novel pathogenic mutations identified in the first Chinese pedigree of complete C6 deficiency.

Authors:  Philip H Li; William Wy Wong; Evelyn Ny Leung; Chak-Sing Lau; Elaine Au
Journal:  Clin Transl Immunology       Date:  2020-07-08

5.  Molecular characterization of invasive capsule null Neisseria meningitidis in South Africa.

Authors:  Karistha Ganesh; Mushal Allam; Nicole Wolter; Holly B Bratcher; Odile B Harrison; Jay Lucidarme; Ray Borrow; Linda de Gouveia; Susan Meiring; Monica Birkhead; Martin C J Maiden; Anne von Gottberg; Mignon du Plessis
Journal:  BMC Microbiol       Date:  2017-02-21       Impact factor: 3.605

6.  Clinical and Genetic Spectrum of a Large Cohort With Total and Sub-total Complement Deficiencies.

Authors:  Carine El Sissy; Jérémie Rosain; Paula Vieira-Martins; Pauline Bordereau; Aurélia Gruber; Magali Devriese; Loïc de Pontual; Muhamed-Kheir Taha; Claire Fieschi; Capucine Picard; Véronique Frémeaux-Bacchi
Journal:  Front Immunol       Date:  2019-08-08       Impact factor: 7.561

7.  Hereditary Deficiency of the Second Component of Complement: Early Diagnosis and 21-Year Follow-Up of a Family.

Authors:  Rosa Maria Dellepiane; Lucia Augusta Baselli; Marco Cazzaniga; Vassilios Lougaris; Paolo Macor; Mara Giordano; Roberta Gualtierotti; Massimo Cugno
Journal:  Medicina (Kaunas)       Date:  2020-03-10       Impact factor: 2.430

8.  Invasive meningococcal disease in three siblings with hereditary deficiency of the 8(th) component of complement: evidence for the importance of an early diagnosis.

Authors:  Rosa Maria Dellepiane; Laura Dell'Era; Paola Pavesi; Paolo Macor; Mara Giordano; Luca De Maso; Maria Cristina Pietrogrande; Massimo Cugno
Journal:  Orphanet J Rare Dis       Date:  2016-05-17       Impact factor: 4.123

9.  Recurrent meningococcal meningitis with complement 6 (C6) deficiency: A case report.

Authors:  Ji Yun Bae; Ahrong Ham; Hee Jung Choi; Chung-Jong Kim
Journal:  Medicine (Baltimore)       Date:  2020-05-22       Impact factor: 1.817

  9 in total

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