C7 deficiency in an Irish family: a deletion defect which is predominant in the Irish

Human deficiencies of terminal complement components are known to be associated with increased susceptibility to Neisseria meningitidis infection. Polymorphic DNA marker studies in complement deficient investigations allow identification of haplotypes associated with the deficiency and enable the po...

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Veröffentlicht in:Clinical and experimental immunology 1998-12, Vol.114 (3), p.355-361
Hauptverfasser: O'HARA, A. M, FERNIE, B. A, MORAN, A. P, WILLIAMS, Y. E, CONNAUGHTON, J. J, ORREN, A, HOBART, M. J
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container_end_page 361
container_issue 3
container_start_page 355
container_title Clinical and experimental immunology
container_volume 114
creator O'HARA, A. M
FERNIE, B. A
MORAN, A. P
WILLIAMS, Y. E
CONNAUGHTON, J. J
ORREN, A
HOBART, M. J
description Human deficiencies of terminal complement components are known to be associated with increased susceptibility to Neisseria meningitidis infection. Polymorphic DNA marker studies in complement deficient investigations allow identification of haplotypes associated with the deficiency and enable the possible identification of heterozygote carriers of the defect. We report studies of an Irish family in which the index case had suffered recurrent meningococcal disease and was found to be deficient in the seventh component of complement (C7). The availability of all family members enabled us to determine the segregating haplotypes. The defects in the family segregated with two very closely related C6 and C7 DNA haplotypes, one of which is known to be associated with the large Irish C7 DNA deletion defect. The index case and two C7 deficient siblings were found to be homozygous for this defect, a deletion that spans approx. 6.8 kbp and encompasses exons 7 and 8. The deletion defect of exons 7 and 8 of C7 has been found in homozygous form in another C7 deficient Irish individual, and is present in heterozygous form in C7 deficient members of a third Irish family. Therefore, this deletion defect occurs in five of the six deficient chromosomes of these three unrelated Irish families, raising the interesting question of how prevalent this defect may be within the Irish community.
doi_str_mv 10.1046/j.1365-2249.1998.00737.x
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The defects in the family segregated with two very closely related C6 and C7 DNA haplotypes, one of which is known to be associated with the large Irish C7 DNA deletion defect. The index case and two C7 deficient siblings were found to be homozygous for this defect, a deletion that spans approx. 6.8 kbp and encompasses exons 7 and 8. The deletion defect of exons 7 and 8 of C7 has been found in homozygous form in another C7 deficient Irish individual, and is present in heterozygous form in C7 deficient members of a third Irish family. 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Immunoglobulinopathies</subject><subject>Immunopathology</subject><subject>Ireland</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Meningococcal Infections - genetics</subject><subject>Meningococcal Infections - immunology</subject><subject>Neisseria meningitidis</subject><subject>Original</subject><subject>Pedigree</subject><subject>Polymerase Chain Reaction</subject><issn>0009-9104</issn><issn>1365-2249</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>1998</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqNUVtr2zAUFmWly7r-hIEYY2_2jmRdrDEGI7RdoLCX9Vkosjwr2HImOWvz7yc3Ibu8bCDQEd_lnKMPIUygJMDEu01JKsELSpkqiVJ1CSArWT6eocUJeIYWAKAKlRXP0YuUNvkphKAX6ELVjAGrFuh-KXHjWm-9C3aPfcAm4FX0qcOtGXy_f49NJvRu8mOYmc5O-KHztsM-4W10zTj4YMI0S6fOHbQv0Xlr-uSujvclur-5_rr8XNx9uV0tP90VlnMui5ZCPlKyNTfOMUqIXHNSNVJyygGokI2pDQcjna3UmtRUGcEgF0IQIVR1iT4efLe79eAa68IUTa-30Q8m7vVovP4TCb7T38YfmijghNJs8PZoEMfvO5cmPfhkXd-b4MZd0hIIBQLkn0QiCSOUi0x8_RdxM-5iyL-Qm4q6Urye564PJBvHlKJrTyMT0HPAeqPnHPWco54D1k8B68csffX7yifhMdGMvzniJlnTt9EE69Mvf65A1fM-Hw60B9-7_X-318vrVS6qn2QQvpM</recordid><startdate>199812</startdate><enddate>199812</enddate><creator>O'HARA, A. 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Therefore, this deletion defect occurs in five of the six deficient chromosomes of these three unrelated Irish families, raising the interesting question of how prevalent this defect may be within the Irish community.</abstract><cop>Oxford BSL</cop><pub>Blackwell Science Ltd</pub><pmid>9844043</pmid><doi>10.1046/j.1365-2249.1998.00737.x</doi><tpages>7</tpages><oa>free_for_read</oa></addata></record>
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ispartof Clinical and experimental immunology, 1998-12, Vol.114 (3), p.355-361
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source Oxford University Press Journals All Titles (1996-Current); MEDLINE; Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals; PubMed Central; Alma/SFX Local Collection
subjects Biological and medical sciences
Blotting, Southern
C7 deficiency
complement
Complement C6 - genetics
Complement C7 - deficiency
Complement C7 - genetics
Complement C7 - immunology
DNA markers
Exons
Female
genetic defects
Genetic Markers
Humans
Immunodeficiencies
Immunodeficiencies. Immunoglobulinopathies
Immunopathology
Ireland
Male
Medical sciences
Meningococcal Infections - genetics
Meningococcal Infections - immunology
Neisseria meningitidis
Original
Pedigree
Polymerase Chain Reaction
title C7 deficiency in an Irish family: a deletion defect which is predominant in the Irish
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