Motor abnormalities in premanifest persons with Huntington's disease: The PREDICT‐HD study
Identifieur interne : 000561 ( Istex/Corpus ); précédent : 000560; suivant : 000562Motor abnormalities in premanifest persons with Huntington's disease: The PREDICT‐HD study
Auteurs : Kevin M. Biglan ; Christopher A. Ross ; Douglas R. Langbehn ; Elizabeth H. Aylward ; Julie C. Stout ; Sarah Queller ; Noelle E. Carlozzi ; Kevin Duff ; Leigh J. Beglinger ; Jane S. PaulsenSource :
- Movement Disorders [ 0885-3185 ] ; 2009-09-15.
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- KwdEn :
Abstract
The PREDICT‐HD study seeks to identify clinical and biological markers of Huntington's disease in premanifest individuals who have undergone predictive genetic testing. We compared baseline motor data between gene‐expansion carriers (cases) and nongene‐expansion carriers (controls) using t‐tests and Chi‐square. Cases were categorized as near, mid, or far from diagnosis using a CAG‐based formula. Striatal volumes were calculated using volumetric magnetic resonance imaging measurements. Multiple linear regression associated total motor score, motor domains, and individual motor items with estimated diagnosis and striatal volumes. Elevated total motor scores at baseline were associated with higher genetic probability of disease diagnosis in the near future (partial R2 0.14, P < 0.0001) and smaller striatal volumes (partial R2 0.15, P < 0.0001). Nearly all motor domain scores showed greater abnormality with increasing proximity to diagnosis, although bradykinesia and chorea were most highly associated with diagnostic immediacy. Among individual motor items, worse scores on finger tapping, tandem gait, Luria, saccade initiation, and chorea show unique association with diagnosis probability. Even in this premanifest population, subtle motor abnormalities were associated with a higher probability of disease diagnosis and smaller striatal volumes. Longitudinal assessment will help inform whether motor items will be useful measures in preventive clinical trials. © 2009 Movement Disorder Society
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DOI: 10.1002/mds.22601
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Biglan</name><affiliation><mods:affiliation>University of Rochester, Rochester, New York, USA</mods:affiliation></affiliation></author><author><name sortKey="Ross, Christopher A" sort="Ross, Christopher A" uniqKey="Ross C" first="Christopher A." last="Ross">Christopher A. Ross</name><affiliation><mods:affiliation>Johns Hopkins University, Baltimore, Maryland, USA</mods:affiliation></affiliation></author><author><name sortKey="Langbehn, Douglas R" sort="Langbehn, Douglas R" uniqKey="Langbehn D" first="Douglas R." last="Langbehn">Douglas R. Langbehn</name><affiliation><mods:affiliation>University of Iowa, Iowa City, Iowa, USA</mods:affiliation></affiliation></author><author><name sortKey="Aylward, Elizabeth H" sort="Aylward, Elizabeth H" uniqKey="Aylward E" first="Elizabeth H." last="Aylward">Elizabeth H. Aylward</name><affiliation><mods:affiliation>University of Washington, Seattle, Washington, USA</mods:affiliation></affiliation></author><author><name sortKey="Stout, Julie C" sort="Stout, Julie C" uniqKey="Stout J" first="Julie C." last="Stout">Julie C. Stout</name><affiliation><mods:affiliation>Monash University, Victoria, Australia</mods:affiliation></affiliation></author><author><name sortKey="Queller, Sarah" sort="Queller, Sarah" uniqKey="Queller S" first="Sarah" last="Queller">Sarah Queller</name><affiliation><mods:affiliation>Indiana University, Bloomington, Indiana, USA</mods:affiliation></affiliation></author><author><name sortKey="Carlozzi, Noelle E" sort="Carlozzi, Noelle E" uniqKey="Carlozzi N" first="Noelle E." last="Carlozzi">Noelle E. Carlozzi</name><affiliation><mods:affiliation>Kessler Medical Rehabilitation Research and Education Center, West Orange, New Jersey, USA</mods:affiliation></affiliation></author><author><name sortKey="Duff, Kevin" sort="Duff, Kevin" uniqKey="Duff K" first="Kevin" last="Duff">Kevin Duff</name><affiliation><mods:affiliation>University of Iowa, Iowa City, Iowa, USA</mods:affiliation></affiliation></author><author><name sortKey="Beglinger, Leigh J" sort="Beglinger, Leigh J" uniqKey="Beglinger L" first="Leigh J." last="Beglinger">Leigh J. Beglinger</name><affiliation><mods:affiliation>University of Iowa, Iowa City, Iowa, USA</mods:affiliation></affiliation></author><author><name sortKey="Paulsen, Jane S" sort="Paulsen, Jane S" uniqKey="Paulsen J" first="Jane S." last="Paulsen">Jane S. Paulsen</name><affiliation><mods:affiliation>University of Iowa, Iowa City, Iowa, USA</mods:affiliation></affiliation></author></analytic><monogr></monogr><series><title level="j">Movement Disorders</title><title level="j" type="abbrev">Mov. Disord.</title><idno type="ISSN">0885-3185</idno><idno type="eISSN">1531-8257</idno><imprint><publisher>Wiley Subscription Services, Inc., A Wiley Company</publisher><pubPlace>Hoboken</pubPlace><date type="published" when="2009-09-15">2009-09-15</date><biblScope unit="vol">24</biblScope><biblScope unit="issue">12</biblScope><biblScope unit="page" from="1763">1763</biblScope><biblScope unit="page" to="1772">1772</biblScope></imprint><idno type="ISSN">0885-3185</idno></series><idno type="istex">6AA2C4AC0C2B0FC427531284AAF17804A28580B2</idno><idno type="DOI">10.1002/mds.22601</idno><idno type="ArticleID">MDS22601</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Huntington's disease</term><term>UHDRS</term><term>at‐risk</term></keywords></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">The PREDICT‐HD study seeks to identify clinical and biological markers of Huntington's disease in premanifest individuals who have undergone predictive genetic testing. We compared baseline motor data between gene‐expansion carriers (cases) and nongene‐expansion carriers (controls) using t‐tests and Chi‐square. Cases were categorized as near, mid, or far from diagnosis using a CAG‐based formula. Striatal volumes were calculated using volumetric magnetic resonance imaging measurements. Multiple linear regression associated total motor score, motor domains, and individual motor items with estimated diagnosis and striatal volumes. Elevated total motor scores at baseline were associated with higher genetic probability of disease diagnosis in the near future (partial R2 0.14, P < 0.0001) and smaller striatal volumes (partial R2 0.15, P < 0.0001). Nearly all motor domain scores showed greater abnormality with increasing proximity to diagnosis, although bradykinesia and chorea were most highly associated with diagnostic immediacy. Among individual motor items, worse scores on finger tapping, tandem gait, Luria, saccade initiation, and chorea show unique association with diagnosis probability. Even in this premanifest population, subtle motor abnormalities were associated with a higher probability of disease diagnosis and smaller striatal volumes. Longitudinal assessment will help inform whether motor items will be useful measures in preventive clinical trials. © 2009 Movement Disorder Society</div></front></TEI><istex><corpusName>wiley</corpusName><author><json:item><name>Kevin M. Biglan MD, MPH</name><affiliations><json:string>University of Rochester, Rochester, New York, USA</json:string></affiliations></json:item><json:item><name>Christopher A. Ross MD, PhD</name><affiliations><json:string>Johns Hopkins University, Baltimore, Maryland, USA</json:string></affiliations></json:item><json:item><name>Douglas R. Langbehn MD, PhD</name><affiliations><json:string>University of Iowa, Iowa City, Iowa, USA</json:string></affiliations></json:item><json:item><name>Elizabeth H. Aylward PhD</name><affiliations><json:string>University of Washington, Seattle, Washington, USA</json:string></affiliations></json:item><json:item><name>Julie C. Stout PhD</name><affiliations><json:string>Monash University, Victoria, Australia</json:string></affiliations></json:item><json:item><name>Sarah Queller PhD</name><affiliations><json:string>Indiana University, Bloomington, Indiana, USA</json:string></affiliations></json:item><json:item><name>Noelle E. Carlozzi PhD</name><affiliations><json:string>Kessler Medical Rehabilitation Research and Education Center, West Orange, New Jersey, USA</json:string></affiliations></json:item><json:item><name>Kevin Duff PhD</name><affiliations><json:string>University of Iowa, Iowa City, Iowa, USA</json:string></affiliations></json:item><json:item><name>Leigh J. Beglinger PhD</name><affiliations><json:string>University of Iowa, Iowa City, Iowa, USA</json:string></affiliations></json:item><json:item><name>Jane S. Paulsen PhD</name><affiliations><json:string>University of Iowa, Iowa City, Iowa, USA</json:string></affiliations></json:item></author><subject><json:item><lang><json:string>eng</json:string></lang><value>Huntington's disease</value></json:item><json:item><lang><json:string>eng</json:string></lang><value>at‐risk</value></json:item><json:item><lang><json:string>eng</json:string></lang><value>UHDRS</value></json:item></subject><language><json:string>eng</json:string></language><abstract>The PREDICT‐HD study seeks to identify clinical and biological markers of Huntington's disease in premanifest individuals who have undergone predictive genetic testing. We compared baseline motor data between gene‐expansion carriers (cases) and nongene‐expansion carriers (controls) using t‐tests and Chi‐square. Cases were categorized as near, mid, or far from diagnosis using a CAG‐based formula. Striatal volumes were calculated using volumetric magnetic resonance imaging measurements. Multiple linear regression associated total motor score, motor domains, and individual motor items with estimated diagnosis and striatal volumes. Elevated total motor scores at baseline were associated with higher genetic probability of disease diagnosis in the near future (partial R2 0.14, P > 0.0001) and smaller striatal volumes (partial R2 0.15, P > 0.0001). Nearly all motor domain scores showed greater abnormality with increasing proximity to diagnosis, although bradykinesia and chorea were most highly associated with diagnostic immediacy. Among individual motor items, worse scores on finger tapping, tandem gait, Luria, saccade initiation, and chorea show unique association with diagnosis probability. Even in this premanifest population, subtle motor abnormalities were associated with a higher probability of disease diagnosis and smaller striatal volumes. Longitudinal assessment will help inform whether motor items will be useful measures in preventive clinical trials. © 2009 Movement Disorder Society</abstract><qualityIndicators><score>7.46</score><pdfVersion>1.3</pdfVersion><pdfPageSize>612 x 810 pts</pdfPageSize><refBibsNative>true</refBibsNative><abstractCharCount>1510</abstractCharCount><pdfWordCount>5289</pdfWordCount><pdfCharCount>36632</pdfCharCount><pdfPageCount>10</pdfPageCount><abstractWordCount>205</abstractWordCount></qualityIndicators><title>Motor abnormalities in premanifest persons with Huntington's disease: The PREDICT‐HD study</title><genre><json:string>Serial article</json:string></genre><host><volume>24</volume><pages><total>10</total><last>1772</last><first>1763</first></pages><issn><json:string>0885-3185</json:string></issn><issue>12</issue><subject><json:item><value>Research Article</value></json:item></subject><genre></genre><language><json:string>unknown</json:string></language><title>Movement Disorders</title><doi><json:string>10.1002/(ISSN)1531-8257</json:string></doi></host><publicationDate>2009</publicationDate><copyrightDate>2009</copyrightDate><doi><json:string>10.1002/mds.22601</json:string></doi><id>6AA2C4AC0C2B0FC427531284AAF17804A28580B2</id><fulltext><json:item><original>true</original><mimetype>application/pdf</mimetype><extension>pdf</extension><uri>https://api.istex.fr/document/6AA2C4AC0C2B0FC427531284AAF17804A28580B2/fulltext/pdf</uri></json:item><json:item><original>false</original><mimetype>application/zip</mimetype><extension>zip</extension><uri>https://api.istex.fr/document/6AA2C4AC0C2B0FC427531284AAF17804A28580B2/fulltext/zip</uri></json:item><istex:fulltextTEI uri="https://api.istex.fr/document/6AA2C4AC0C2B0FC427531284AAF17804A28580B2/fulltext/tei"><teiHeader type="text"><fileDesc><titleStmt><title level="a" type="main" xml:lang="en">Motor abnormalities in premanifest persons with Huntington's disease: The PREDICT‐HD study</title></titleStmt><publicationStmt><authority>ISTEX</authority><publisher>Wiley Subscription Services, Inc., A Wiley Company</publisher><pubPlace>Hoboken</pubPlace><availability><p>Wiley Subscription Services, Inc., A Wiley Company</p></availability><date>2009</date></publicationStmt><notesStmt><note type="content">*Potential conflict of interest: Nothing to report.</note><note>National Institutes of Health - No. NS40068;</note><note>CHDI, Inc</note><note>Douglas Langbehn at The University of Iowa</note></notesStmt><sourceDesc><biblStruct type="inbook"><analytic><title level="a" type="main" xml:lang="en">Motor abnormalities in premanifest persons with Huntington's disease: The PREDICT‐HD study</title><author><persName><forename type="first">Kevin M.</forename><surname>Biglan</surname><roleName type="degree">MD, MPH</roleName></persName><affiliation>University of Rochester, Rochester, New York, USA</affiliation></author><author><persName><forename type="first">Christopher A.</forename><surname>Ross</surname><roleName type="degree">MD, PhD</roleName></persName><affiliation>Johns Hopkins University, Baltimore, Maryland, USA</affiliation></author><author><persName><forename type="first">Douglas R.</forename><surname>Langbehn</surname><roleName type="degree">MD, PhD</roleName></persName><affiliation>University of Iowa, Iowa City, Iowa, USA</affiliation></author><author><persName><forename type="first">Elizabeth H.</forename><surname>Aylward</surname><roleName type="degree">PhD</roleName></persName><affiliation>University of Washington, Seattle, Washington, USA</affiliation></author><author><persName><forename type="first">Julie C.</forename><surname>Stout</surname><roleName type="degree">PhD</roleName></persName><affiliation>Monash University, Victoria, Australia</affiliation></author><author><persName><forename type="first">Sarah</forename><surname>Queller</surname><roleName type="degree">PhD</roleName></persName><affiliation>Indiana University, Bloomington, Indiana, USA</affiliation></author><author><persName><forename type="first">Noelle E.</forename><surname>Carlozzi</surname><roleName type="degree">PhD</roleName></persName><affiliation>Kessler Medical Rehabilitation Research and Education Center, West Orange, New Jersey, USA</affiliation></author><author><persName><forename type="first">Kevin</forename><surname>Duff</surname><roleName type="degree">PhD</roleName></persName><affiliation>University of Iowa, Iowa City, Iowa, USA</affiliation></author><author><persName><forename type="first">Leigh J.</forename><surname>Beglinger</surname><roleName type="degree">PhD</roleName></persName><affiliation>University of Iowa, Iowa City, Iowa, USA</affiliation></author><author><persName><forename type="first">Jane S.</forename><surname>Paulsen</surname><roleName type="degree">PhD</roleName></persName><note type="correspondence"><p>Correspondence: University of Iowa, College of Medicine, 1‐305 MEB, Iowa City, IA 52242</p></note><affiliation>University of Iowa, Iowa City, Iowa, USA</affiliation></author></analytic><monogr><title level="j">Movement Disorders</title><title level="j" type="abbrev">Mov. 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We compared baseline motor data between gene‐expansion carriers (cases) and nongene‐expansion carriers (controls) using t‐tests and Chi‐square. Cases were categorized as near, mid, or far from diagnosis using a CAG‐based formula. Striatal volumes were calculated using volumetric magnetic resonance imaging measurements. Multiple linear regression associated total motor score, motor domains, and individual motor items with estimated diagnosis and striatal volumes. Elevated total motor scores at baseline were associated with higher genetic probability of disease diagnosis in the near future (partial R2 0.14, P < 0.0001) and smaller striatal volumes (partial R2 0.15, P < 0.0001). Nearly all motor domain scores showed greater abnormality with increasing proximity to diagnosis, although bradykinesia and chorea were most highly associated with diagnostic immediacy. Among individual motor items, worse scores on finger tapping, tandem gait, Luria, saccade initiation, and chorea show unique association with diagnosis probability. Even in this premanifest population, subtle motor abnormalities were associated with a higher probability of disease diagnosis and smaller striatal volumes. Longitudinal assessment will help inform whether motor items will be useful measures in preventive clinical trials. © 2009 Movement Disorder Society</p></abstract><textClass xml:lang="en"><keywords scheme="keyword"><list><head>Keywords</head><item><term>Huntington's disease</term></item><item><term>at‐risk</term></item><item><term>UHDRS</term></item></list></keywords></textClass><textClass><keywords scheme="Journal Subject"><list><head>Article category</head><item><term>Research Article</term></item></list></keywords></textClass></profileDesc><revisionDesc><change when="2008-12-09">Received</change><change when="2009-03-17">Registration</change><change when="2009-09-15">Published</change></revisionDesc></teiHeader></istex:fulltextTEI><json:item><original>false</original><mimetype>text/plain</mimetype><extension>txt</extension><uri>https://api.istex.fr/document/6AA2C4AC0C2B0FC427531284AAF17804A28580B2/fulltext/txt</uri></json:item></fulltext><metadata><istex:metadataXml wicri:clean="Wiley, elements deleted: body"><istex:xmlDeclaration>version="1.0" encoding="UTF-8" standalone="yes"</istex:xmlDeclaration><istex:document><component version="2.0" type="serialArticle" xml:lang="en"><header><publicationMeta level="product"><publisherInfo><publisherName>Wiley Subscription Services, Inc., A Wiley Company</publisherName><publisherLoc>Hoboken</publisherLoc></publisherInfo><doi registered="yes">10.1002/(ISSN)1531-8257</doi><issn type="print">0885-3185</issn><issn type="electronic">1531-8257</issn><idGroup><id type="product" value="MDS"></id></idGroup><titleGroup><title type="main" xml:lang="en" sort="MOVEMENT DISORDERS">Movement Disorders</title><title type="short">Mov. 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affiliationRef="#af3"><personName><givenNames>Douglas R.</givenNames><familyName>Langbehn</familyName><degrees>MD, PhD</degrees></personName></creator><creator xml:id="au4" creatorRole="author" affiliationRef="#af4"><personName><givenNames>Elizabeth H.</givenNames><familyName>Aylward</familyName><degrees>PhD</degrees></personName></creator><creator xml:id="au5" creatorRole="author" affiliationRef="#af5"><personName><givenNames>Julie C.</givenNames><familyName>Stout</familyName><degrees>PhD</degrees></personName></creator><creator xml:id="au6" creatorRole="author" affiliationRef="#af6"><personName><givenNames>Sarah</givenNames><familyName>Queller</familyName><degrees>PhD</degrees></personName></creator><creator xml:id="au7" creatorRole="author" affiliationRef="#af7"><personName><givenNames>Noelle E.</givenNames><familyName>Carlozzi</familyName><degrees>PhD</degrees></personName></creator><creator xml:id="au8" creatorRole="author" affiliationRef="#af3"><personName><givenNames>Kevin</givenNames><familyName>Duff</familyName><degrees>PhD</degrees></personName></creator><creator xml:id="au9" creatorRole="author" affiliationRef="#af3"><personName><givenNames>Leigh J.</givenNames><familyName>Beglinger</familyName><degrees>PhD</degrees></personName></creator><creator xml:id="au10" creatorRole="author" affiliationRef="#af3" corresponding="yes"><personName><givenNames>Jane S.</givenNames><familyName>Paulsen</familyName><degrees>PhD</degrees></personName><contactDetails><email normalForm="jane-paulsen@uiowa.edu">jane‐paulsen@uiowa.edu</email></contactDetails></creator></creators><affiliationGroup><affiliation xml:id="af1" countryCode="US" type="organization"><unparsedAffiliation>University of Rochester, Rochester, New York, USA</unparsedAffiliation></affiliation><affiliation xml:id="af2" countryCode="US" type="organization"><unparsedAffiliation>Johns Hopkins University, Baltimore, Maryland, USA</unparsedAffiliation></affiliation><affiliation xml:id="af3" countryCode="US" type="organization"><unparsedAffiliation>University of Iowa, Iowa City, Iowa, USA</unparsedAffiliation></affiliation><affiliation xml:id="af4" countryCode="US" type="organization"><unparsedAffiliation>University of Washington, Seattle, Washington, USA</unparsedAffiliation></affiliation><affiliation xml:id="af5" countryCode="AU" type="organization"><unparsedAffiliation>Monash University, Victoria, Australia</unparsedAffiliation></affiliation><affiliation xml:id="af6" countryCode="US" type="organization"><unparsedAffiliation>Indiana University, Bloomington, Indiana, USA</unparsedAffiliation></affiliation><affiliation xml:id="af7" countryCode="US" type="organization"><unparsedAffiliation>Kessler Medical Rehabilitation Research and Education Center, West Orange, New Jersey, USA</unparsedAffiliation></affiliation></affiliationGroup><keywordGroup xml:lang="en" type="author"><keyword xml:id="kwd1">Huntington's disease</keyword><keyword xml:id="kwd2">at‐risk</keyword><keyword xml:id="kwd3">UHDRS</keyword></keywordGroup><fundingInfo><fundingAgency>National Institutes of Health</fundingAgency><fundingNumber>NS40068</fundingNumber></fundingInfo><fundingInfo><fundingAgency>CHDI, Inc</fundingAgency></fundingInfo><fundingInfo><fundingAgency>Douglas Langbehn at The University of Iowa</fundingAgency></fundingInfo><abstractGroup><abstract type="main" xml:lang="en"><title type="main">Abstract</title><p>The PREDICT‐HD study seeks to identify clinical and biological markers of Huntington's disease in premanifest individuals who have undergone predictive genetic testing. We compared baseline motor data between gene‐expansion carriers (cases) and nongene‐expansion carriers (controls) using <i>t</i>‐tests and Chi‐square. Cases were categorized as near, mid, or far from diagnosis using a CAG‐based formula. Striatal volumes were calculated using volumetric magnetic resonance imaging measurements. Multiple linear regression associated total motor score, motor domains, and individual motor items with estimated diagnosis and striatal volumes. Elevated total motor scores at baseline were associated with higher genetic probability of disease diagnosis in the near future (partial R<sup>2</sup> 0.14, <i>P</i> < 0.0001) and smaller striatal volumes (partial R<sup>2</sup> 0.15, <i>P</i> < 0.0001). Nearly all motor domain scores showed greater abnormality with increasing proximity to diagnosis, although bradykinesia and chorea were most highly associated with diagnostic immediacy. Among individual motor items, worse scores on finger tapping, tandem gait, Luria, saccade initiation, and chorea show unique association with diagnosis probability. Even in this premanifest population, subtle motor abnormalities were associated with a higher probability of disease diagnosis and smaller striatal volumes. Longitudinal assessment will help inform whether motor items will be useful measures in preventive clinical trials. © 2009 Movement Disorder Society</p></abstract></abstractGroup></contentMeta><noteGroup><note xml:id="fn2"><p>Potential conflict of interest: Nothing to report.</p></note></noteGroup></header></component></istex:document></istex:metadataXml><!--Version 0.6 générée le 4-12-2015--><mods version="3.6"><titleInfo lang="en"><title>Motor abnormalities in premanifest persons with Huntington's disease: The PREDICT‐HD study</title></titleInfo><titleInfo type="abbreviated" lang="en"><title>Motor Abnormalities in Premanifest HD</title></titleInfo><titleInfo type="alternative" contentType="CDATA" lang="en"><title>Motor abnormalities in premanifest persons with Huntington's disease: The PREDICT‐HD study</title></titleInfo><name type="personal"><namePart type="given">Kevin M.</namePart><namePart type="family">Biglan</namePart><namePart type="termsOfAddress">MD, MPH</namePart><affiliation>University of Rochester, Rochester, New York, USA</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Christopher A.</namePart><namePart type="family">Ross</namePart><namePart type="termsOfAddress">MD, PhD</namePart><affiliation>Johns Hopkins University, Baltimore, Maryland, USA</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Douglas R.</namePart><namePart type="family">Langbehn</namePart><namePart type="termsOfAddress">MD, PhD</namePart><affiliation>University of Iowa, Iowa City, Iowa, USA</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Elizabeth H.</namePart><namePart type="family">Aylward</namePart><namePart type="termsOfAddress">PhD</namePart><affiliation>University of Washington, Seattle, Washington, USA</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Julie C.</namePart><namePart type="family">Stout</namePart><namePart type="termsOfAddress">PhD</namePart><affiliation>Monash University, Victoria, Australia</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Sarah</namePart><namePart type="family">Queller</namePart><namePart type="termsOfAddress">PhD</namePart><affiliation>Indiana University, Bloomington, Indiana, USA</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Noelle E.</namePart><namePart type="family">Carlozzi</namePart><namePart type="termsOfAddress">PhD</namePart><affiliation>Kessler Medical Rehabilitation Research and Education Center, West Orange, New Jersey, USA</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Kevin</namePart><namePart type="family">Duff</namePart><namePart type="termsOfAddress">PhD</namePart><affiliation>University of Iowa, Iowa City, Iowa, USA</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Leigh J.</namePart><namePart type="family">Beglinger</namePart><namePart type="termsOfAddress">PhD</namePart><affiliation>University of Iowa, Iowa City, Iowa, USA</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Jane S.</namePart><namePart type="family">Paulsen</namePart><namePart type="termsOfAddress">PhD</namePart><affiliation>University of Iowa, Iowa City, Iowa, USA</affiliation><description>Correspondence: University of Iowa, College of Medicine, 1‐305 MEB, Iowa City, IA 52242</description><role><roleTerm type="text">author</roleTerm></role></name><typeOfResource>text</typeOfResource><genre authority="originalCategForm">article</genre><originInfo><publisher>Wiley Subscription Services, Inc., A Wiley Company</publisher><place><placeTerm type="text">Hoboken</placeTerm></place><dateIssued encoding="w3cdtf">2009-09-15</dateIssued><dateCaptured encoding="w3cdtf">2008-12-09</dateCaptured><dateValid encoding="w3cdtf">2009-03-17</dateValid><copyrightDate encoding="w3cdtf">2009</copyrightDate></originInfo><language><languageTerm type="code" authority="rfc3066">en</languageTerm><languageTerm type="code" authority="iso639-2b">eng</languageTerm></language><physicalDescription><internetMediaType>text/html</internetMediaType><extent unit="figures">1</extent><extent unit="tables">4</extent><extent unit="references">33</extent><extent unit="words">7184</extent></physicalDescription><abstract lang="en">The PREDICT‐HD study seeks to identify clinical and biological markers of Huntington's disease in premanifest individuals who have undergone predictive genetic testing. We compared baseline motor data between gene‐expansion carriers (cases) and nongene‐expansion carriers (controls) using t‐tests and Chi‐square. Cases were categorized as near, mid, or far from diagnosis using a CAG‐based formula. Striatal volumes were calculated using volumetric magnetic resonance imaging measurements. Multiple linear regression associated total motor score, motor domains, and individual motor items with estimated diagnosis and striatal volumes. Elevated total motor scores at baseline were associated with higher genetic probability of disease diagnosis in the near future (partial R2 0.14, P < 0.0001) and smaller striatal volumes (partial R2 0.15, P < 0.0001). Nearly all motor domain scores showed greater abnormality with increasing proximity to diagnosis, although bradykinesia and chorea were most highly associated with diagnostic immediacy. Among individual motor items, worse scores on finger tapping, tandem gait, Luria, saccade initiation, and chorea show unique association with diagnosis probability. Even in this premanifest population, subtle motor abnormalities were associated with a higher probability of disease diagnosis and smaller striatal volumes. Longitudinal assessment will help inform whether motor items will be useful measures in preventive clinical trials. © 2009 Movement Disorder Society</abstract><note type="content">*Potential conflict of interest: Nothing to report.</note><note type="funding">National Institutes of Health - No. NS40068; </note><note type="funding">CHDI, Inc</note><note type="funding">Douglas Langbehn at The University of Iowa</note><subject lang="en"><genre>Keywords</genre><topic>Huntington's disease</topic><topic>at‐risk</topic><topic>UHDRS</topic></subject><relatedItem type="host"><titleInfo><title>Movement Disorders</title></titleInfo><titleInfo type="abbreviated"><title>Mov. Disord.</title></titleInfo><subject><genre>article category</genre><topic>Research Article</topic></subject><identifier type="ISSN">0885-3185</identifier><identifier type="eISSN">1531-8257</identifier><identifier type="DOI">10.1002/(ISSN)1531-8257</identifier><identifier type="PublisherID">MDS</identifier><part><date>2009</date><detail type="volume"><caption>vol.</caption><number>24</number></detail><detail type="issue"><caption>no.</caption><number>12</number></detail><extent unit="pages"><start>1763</start><end>1772</end><total>10</total></extent></part></relatedItem><identifier type="istex">6AA2C4AC0C2B0FC427531284AAF17804A28580B2</identifier><identifier type="DOI">10.1002/mds.22601</identifier><identifier type="ArticleID">MDS22601</identifier><accessCondition type="use and reproduction" contentType="copyright">Copyright © 2009 Movement Disorder Society</accessCondition><recordInfo><recordOrigin>Wiley Subscription Services, Inc., A Wiley Company</recordOrigin><recordContentSource>WILEY</recordContentSource></recordInfo></mods></metadata><serie></serie></istex></record>Pour manipuler ce document sous Unix (Dilib)
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