Clinical findings of a myoclonus-dystonia family with two distinct mutations
Myoclonus-dystonia has recently been associated with mutations in the epsilon-sarcoglycan gene (SCGE) on 7q21. Previously, the authors reported a patient with myoclonus-dystonia and an 18-bp deletion in the DYT1 gene on 9q34. The authors have now re-evaluated the patient harboring this deletion for...
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Veröffentlicht in: | Neurology 2002-10, Vol.59 (8), p.1244-1246 |
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creator | DOHENY, D DANISI, F BREAKEFIELD, X. O BRIN, M. F SILVERMAN, J. M SMITH, C MORRISON, C VELICKOVIC, M DE LEON, D BRESSMAN, S. B LEUNG, J OZELIUS, L KLEIN, C |
description | Myoclonus-dystonia has recently been associated with mutations in the epsilon-sarcoglycan gene (SCGE) on 7q21. Previously, the authors reported a patient with myoclonus-dystonia and an 18-bp deletion in the DYT1 gene on 9q34. The authors have now re-evaluated the patient harboring this deletion for mutations in the SGCE gene and identified a missense change. In the current study, the authors describe the clinical details of this family carrying mutations in two different dystonia genes. Further analysis of these mutations separately and together in cell culture and in animal models should clarify their functional consequences. |
doi_str_mv | 10.1212/WNL.59.8.1244 |
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O ; BRIN, M. F ; SILVERMAN, J. M ; SMITH, C ; MORRISON, C ; VELICKOVIC, M ; DE LEON, D ; BRESSMAN, S. B ; LEUNG, J ; OZELIUS, L ; KLEIN, C</creator><creatorcontrib>DOHENY, D ; DANISI, F ; BREAKEFIELD, X. O ; BRIN, M. F ; SILVERMAN, J. M ; SMITH, C ; MORRISON, C ; VELICKOVIC, M ; DE LEON, D ; BRESSMAN, S. B ; LEUNG, J ; OZELIUS, L ; KLEIN, C</creatorcontrib><description>Myoclonus-dystonia has recently been associated with mutations in the epsilon-sarcoglycan gene (SCGE) on 7q21. Previously, the authors reported a patient with myoclonus-dystonia and an 18-bp deletion in the DYT1 gene on 9q34. The authors have now re-evaluated the patient harboring this deletion for mutations in the SGCE gene and identified a missense change. In the current study, the authors describe the clinical details of this family carrying mutations in two different dystonia genes. Further analysis of these mutations separately and together in cell culture and in animal models should clarify their functional consequences.</description><identifier>ISSN: 0028-3878</identifier><identifier>EISSN: 1526-632X</identifier><identifier>DOI: 10.1212/WNL.59.8.1244</identifier><identifier>PMID: 12391355</identifier><identifier>CODEN: NEURAI</identifier><language>eng</language><publisher>Hagerstown, MD: Lippincott Williams & Wilkins</publisher><subject>Adolescent ; Biological and medical sciences ; Carrier Proteins - genetics ; Cytoskeletal Proteins - genetics ; Dystonia - genetics ; Dystonia - psychology ; Female ; Humans ; Male ; Medical sciences ; Membrane Glycoproteins - genetics ; Middle Aged ; Molecular Chaperones ; Mutation - genetics ; Myoclonus - genetics ; Myoclonus - psychology ; Nervous system (semeiology, syndromes) ; Nervous system as a whole ; Neurology ; Neuropsychological Tests ; Pedigree ; Sarcoglycans</subject><ispartof>Neurology, 2002-10, Vol.59 (8), p.1244-1246</ispartof><rights>2003 INIST-CNRS</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c319t-2eb18cf48f9e0bed6792f751c6175146a84854d624d5bd31eea405043d68915a3</citedby><cites>FETCH-LOGICAL-c319t-2eb18cf48f9e0bed6792f751c6175146a84854d624d5bd31eea405043d68915a3</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,776,780,27901,27902</link.rule.ids><backlink>$$Uhttp://pascal-francis.inist.fr/vibad/index.php?action=getRecordDetail&idt=14444864$$DView record in Pascal Francis$$Hfree_for_read</backlink><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/12391355$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>DOHENY, D</creatorcontrib><creatorcontrib>DANISI, F</creatorcontrib><creatorcontrib>BREAKEFIELD, X. 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In the current study, the authors describe the clinical details of this family carrying mutations in two different dystonia genes. Further analysis of these mutations separately and together in cell culture and in animal models should clarify their functional consequences.</description><subject>Adolescent</subject><subject>Biological and medical sciences</subject><subject>Carrier Proteins - genetics</subject><subject>Cytoskeletal Proteins - genetics</subject><subject>Dystonia - genetics</subject><subject>Dystonia - psychology</subject><subject>Female</subject><subject>Humans</subject><subject>Male</subject><subject>Medical sciences</subject><subject>Membrane Glycoproteins - genetics</subject><subject>Middle Aged</subject><subject>Molecular Chaperones</subject><subject>Mutation - genetics</subject><subject>Myoclonus - genetics</subject><subject>Myoclonus - psychology</subject><subject>Nervous system (semeiology, syndromes)</subject><subject>Nervous system as a whole</subject><subject>Neurology</subject><subject>Neuropsychological Tests</subject><subject>Pedigree</subject><subject>Sarcoglycans</subject><issn>0028-3878</issn><issn>1526-632X</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2002</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNpFkDtLBDEURoMo7voobSWNdrPmOZOUsviCRRtFu5DNQyMziU4yyP57s7jgLe7l4x6-4gBwhtECE0yuXh9XCy4XoibG9sAcc9I2LSVv-2COEBENFZ2YgaOcPxGqz04eghkmVGLK-Rysln2Iwege-hBtiO8ZJg81HDbJ9ClOubGbXFIMGno9hH4Df0L5gOUnQRtyCdEUOExFl5BiPgEHXvfZne7uMXi5vXle3jerp7uH5fWqMRTL0hC3xsJ4Jrx0aO1s20niO45Ni-tmrRZMcGZbwixfW4qd0wxxxKhthcRc02Nw-df7NabvyeWihpCN63sdXZqy6giWHGNZweYPNGPKeXRefY1h0ONGYaS2-lTVp7hUQm31Vf58VzytB2f_6Z2vClzsAJ2rND_qaEL-51gd0TL6CwvKd-Q</recordid><startdate>20021022</startdate><enddate>20021022</enddate><creator>DOHENY, D</creator><creator>DANISI, F</creator><creator>BREAKEFIELD, X. 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F</au><au>SILVERMAN, J. M</au><au>SMITH, C</au><au>MORRISON, C</au><au>VELICKOVIC, M</au><au>DE LEON, D</au><au>BRESSMAN, S. B</au><au>LEUNG, J</au><au>OZELIUS, L</au><au>KLEIN, C</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Clinical findings of a myoclonus-dystonia family with two distinct mutations</atitle><jtitle>Neurology</jtitle><addtitle>Neurology</addtitle><date>2002-10-22</date><risdate>2002</risdate><volume>59</volume><issue>8</issue><spage>1244</spage><epage>1246</epage><pages>1244-1246</pages><issn>0028-3878</issn><eissn>1526-632X</eissn><coden>NEURAI</coden><abstract>Myoclonus-dystonia has recently been associated with mutations in the epsilon-sarcoglycan gene (SCGE) on 7q21. Previously, the authors reported a patient with myoclonus-dystonia and an 18-bp deletion in the DYT1 gene on 9q34. The authors have now re-evaluated the patient harboring this deletion for mutations in the SGCE gene and identified a missense change. In the current study, the authors describe the clinical details of this family carrying mutations in two different dystonia genes. Further analysis of these mutations separately and together in cell culture and in animal models should clarify their functional consequences.</abstract><cop>Hagerstown, MD</cop><pub>Lippincott Williams & Wilkins</pub><pmid>12391355</pmid><doi>10.1212/WNL.59.8.1244</doi><tpages>3</tpages></addata></record> |
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subjects | Adolescent Biological and medical sciences Carrier Proteins - genetics Cytoskeletal Proteins - genetics Dystonia - genetics Dystonia - psychology Female Humans Male Medical sciences Membrane Glycoproteins - genetics Middle Aged Molecular Chaperones Mutation - genetics Myoclonus - genetics Myoclonus - psychology Nervous system (semeiology, syndromes) Nervous system as a whole Neurology Neuropsychological Tests Pedigree Sarcoglycans |
title | Clinical findings of a myoclonus-dystonia family with two distinct mutations |
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