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 |
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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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M ; FERNIE, B. A ; MORAN, A. P ; WILLIAMS, Y. E ; CONNAUGHTON, J. J ; ORREN, A ; HOBART, M. J</creator><creatorcontrib>O'HARA, A. M ; FERNIE, B. A ; MORAN, A. P ; WILLIAMS, Y. E ; CONNAUGHTON, J. J ; ORREN, A ; HOBART, M. J</creatorcontrib><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.</description><identifier>ISSN: 0009-9104</identifier><identifier>EISSN: 1365-2249</identifier><identifier>DOI: 10.1046/j.1365-2249.1998.00737.x</identifier><identifier>PMID: 9844043</identifier><identifier>CODEN: CEXIAL</identifier><language>eng</language><publisher>Oxford BSL: Blackwell Science Ltd</publisher><subject>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</subject><ispartof>Clinical and experimental immunology, 1998-12, Vol.114 (3), p.355-361</ispartof><rights>Blackwell Science Ltd, Oxford</rights><rights>1999 INIST-CNRS</rights><rights>Copyright Blackwell Scientific Publications Ltd. Dec 1998</rights><rights>1998 Blackwell Science Ltd 1998</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c5557-f20f20774b5aee42117b513d7752500267da8a50a7ec39b1829a640b186616693</citedby><cites>FETCH-LOGICAL-c5557-f20f20774b5aee42117b513d7752500267da8a50a7ec39b1829a640b186616693</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC1905122/pdf/$$EPDF$$P50$$Gpubmedcentral$$H</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC1905122/$$EHTML$$P50$$Gpubmedcentral$$H</linktohtml><link.rule.ids>230,314,723,776,780,881,27901,27902,53766,53768</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=1590981$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/9844043$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>O'HARA, A. M</creatorcontrib><creatorcontrib>FERNIE, B. A</creatorcontrib><creatorcontrib>MORAN, A. P</creatorcontrib><creatorcontrib>WILLIAMS, Y. E</creatorcontrib><creatorcontrib>CONNAUGHTON, J. J</creatorcontrib><creatorcontrib>ORREN, A</creatorcontrib><creatorcontrib>HOBART, M. J</creatorcontrib><title>C7 deficiency in an Irish family: a deletion defect which is predominant in the Irish</title><title>Clinical and experimental immunology</title><addtitle>Clin Exp Immunol</addtitle><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.</description><subject>Biological and medical sciences</subject><subject>Blotting, Southern</subject><subject>C7 deficiency</subject><subject>complement</subject><subject>Complement C6 - genetics</subject><subject>Complement C7 - deficiency</subject><subject>Complement C7 - genetics</subject><subject>Complement C7 - immunology</subject><subject>DNA markers</subject><subject>Exons</subject><subject>Female</subject><subject>genetic defects</subject><subject>Genetic Markers</subject><subject>Humans</subject><subject>Immunodeficiencies</subject><subject>Immunodeficiencies. 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. M</creator><creator>FERNIE, B. A</creator><creator>MORAN, A. P</creator><creator>WILLIAMS, Y. E</creator><creator>CONNAUGHTON, J. J</creator><creator>ORREN, A</creator><creator>HOBART, M. J</creator><general>Blackwell Science Ltd</general><general>Blackwell</general><general>Oxford University Press</general><general>Blackwell Publishing Ltd</general><scope>IQODW</scope><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7T5</scope><scope>7U9</scope><scope>H94</scope><scope>M7N</scope><scope>7X8</scope><scope>5PM</scope></search><sort><creationdate>199812</creationdate><title>C7 deficiency in an Irish family: a deletion defect which is predominant in the Irish</title><author>O'HARA, A. M ; FERNIE, B. A ; MORAN, A. P ; WILLIAMS, Y. E ; CONNAUGHTON, J. J ; ORREN, A ; HOBART, M. J</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c5557-f20f20774b5aee42117b513d7752500267da8a50a7ec39b1829a640b186616693</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>1998</creationdate><topic>Biological and medical sciences</topic><topic>Blotting, Southern</topic><topic>C7 deficiency</topic><topic>complement</topic><topic>Complement C6 - genetics</topic><topic>Complement C7 - deficiency</topic><topic>Complement C7 - genetics</topic><topic>Complement C7 - immunology</topic><topic>DNA markers</topic><topic>Exons</topic><topic>Female</topic><topic>genetic defects</topic><topic>Genetic Markers</topic><topic>Humans</topic><topic>Immunodeficiencies</topic><topic>Immunodeficiencies. Immunoglobulinopathies</topic><topic>Immunopathology</topic><topic>Ireland</topic><topic>Male</topic><topic>Medical sciences</topic><topic>Meningococcal Infections - genetics</topic><topic>Meningococcal Infections - immunology</topic><topic>Neisseria meningitidis</topic><topic>Original</topic><topic>Pedigree</topic><topic>Polymerase Chain Reaction</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>O'HARA, A. M</creatorcontrib><creatorcontrib>FERNIE, B. A</creatorcontrib><creatorcontrib>MORAN, A. P</creatorcontrib><creatorcontrib>WILLIAMS, Y. E</creatorcontrib><creatorcontrib>CONNAUGHTON, J. J</creatorcontrib><creatorcontrib>ORREN, A</creatorcontrib><creatorcontrib>HOBART, M. J</creatorcontrib><collection>Pascal-Francis</collection><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>Immunology Abstracts</collection><collection>Virology and AIDS Abstracts</collection><collection>AIDS and Cancer Research Abstracts</collection><collection>Algology Mycology and Protozoology Abstracts (Microbiology C)</collection><collection>MEDLINE - Academic</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Clinical and experimental immunology</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>O'HARA, A. M</au><au>FERNIE, B. A</au><au>MORAN, A. P</au><au>WILLIAMS, Y. E</au><au>CONNAUGHTON, J. J</au><au>ORREN, A</au><au>HOBART, M. J</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>C7 deficiency in an Irish family: a deletion defect which is predominant in the Irish</atitle><jtitle>Clinical and experimental immunology</jtitle><addtitle>Clin Exp Immunol</addtitle><date>1998-12</date><risdate>1998</risdate><volume>114</volume><issue>3</issue><spage>355</spage><epage>361</epage><pages>355-361</pages><issn>0009-9104</issn><eissn>1365-2249</eissn><coden>CEXIAL</coden><abstract>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.</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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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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