Tongue force analysis assesses motor phenotype in premanifest and symptomatic Huntington's disease
Identifieur interne : 000D80 ( Istex/Corpus ); précédent : 000D79; suivant : 000D81Tongue force analysis assesses motor phenotype in premanifest and symptomatic Huntington's disease
Auteurs : Ralf Reilmann ; Stefan Bohlen ; Thomas Klopstock ; Andreas Bender ; Adolf Weindl ; Philipp Saemann ; Dorothee P. Auer ; E. Bernd Ringelstein ; Herwig W. LangeSource :
- Movement Disorders [ 0885-3185 ] ; 2010-10-15.
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- KwdEn :
Abstract
Motor symptoms in Huntington's Disease (HD) are commonly assessed by the Unified Huntington's Disease Rating Scale‐Total Motor Score (UHDRS‐TMS). However, the UHDRS‐TMS is limited by interrater variability, its categorical nature, and insensitivity in premanifest subjects. More objective and quantitative measures of motor phenotype may complement the use of the UHDRS‐TMS as outcome measure and increase the power and sensitivity of clinical trials. Deficits in tongue protrusion are well acknowledged in HD and constitute a subitem of the UHDRS‐TMS. We, therefore, investigated whether objective and quantitative assessment of tongue protrusion forces (TPF) provides measures that (1) correlate to the severity of motor phenotype detected in the UHDRS‐TMS in symptomatic HD, (2) detect a motor phenotype in premanifest HD gene‐carriers, and (3) exhibit a correlation to the genotype as assessed by a disease burden score (based on CAG‐repeat length and age). Using a precalibrated force transducer, the ability of premanifest gene carriers (n = 15) and subjects with symptomatic HD (n = 20) to generate and maintain isometric TPF at three target force levels (0.25, 0.5, and 1.0 N) was assessed and compared with age‐matched controls (n = 20) in a cross‐sectional study. Measures of variability of TPF and tongue contact time distinguished controls, premanifest, and symptomatic HD groups and correlated to the UHDRS‐TMS and disease burden score, suggesting a strong genotype‐phenotype correlation. Group distinction was most reliable at the lowest target force level. We conclude that assessment of TPF may be a useful objective and quantitative marker of motor dysfunction in premanifest and symptomatic HD. © 2010 Movement Disorder Society
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DOI: 10.1002/mds.23243
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Auer</name><affiliation><mods:affiliation>Department of Radiology, Max Planck Institute of Psychiatry, Munich, Germany</mods:affiliation></affiliation></author><author><name sortKey="Ringelstein, E Bernd" sort="Ringelstein, E Bernd" uniqKey="Ringelstein E" first="E. Bernd" last="Ringelstein">E. Bernd Ringelstein</name><affiliation><mods:affiliation>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</mods:affiliation></affiliation></author><author><name sortKey="Lange, Herwig W" sort="Lange, Herwig W" uniqKey="Lange H" first="Herwig W." last="Lange">Herwig W. Lange</name><affiliation><mods:affiliation>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</mods:affiliation></affiliation></author></analytic><monogr></monogr><series><title level="j">Movement Disorders</title><title level="j" type="abbrev">Mov. 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However, the UHDRS‐TMS is limited by interrater variability, its categorical nature, and insensitivity in premanifest subjects. More objective and quantitative measures of motor phenotype may complement the use of the UHDRS‐TMS as outcome measure and increase the power and sensitivity of clinical trials. Deficits in tongue protrusion are well acknowledged in HD and constitute a subitem of the UHDRS‐TMS. We, therefore, investigated whether objective and quantitative assessment of tongue protrusion forces (TPF) provides measures that (1) correlate to the severity of motor phenotype detected in the UHDRS‐TMS in symptomatic HD, (2) detect a motor phenotype in premanifest HD gene‐carriers, and (3) exhibit a correlation to the genotype as assessed by a disease burden score (based on CAG‐repeat length and age). Using a precalibrated force transducer, the ability of premanifest gene carriers (n = 15) and subjects with symptomatic HD (n = 20) to generate and maintain isometric TPF at three target force levels (0.25, 0.5, and 1.0 N) was assessed and compared with age‐matched controls (n = 20) in a cross‐sectional study. Measures of variability of TPF and tongue contact time distinguished controls, premanifest, and symptomatic HD groups and correlated to the UHDRS‐TMS and disease burden score, suggesting a strong genotype‐phenotype correlation. Group distinction was most reliable at the lowest target force level. We conclude that assessment of TPF may be a useful objective and quantitative marker of motor dysfunction in premanifest and symptomatic HD. © 2010 Movement Disorder Society</div></front></TEI><istex><corpusName>wiley</corpusName><author><json:item><name>Ralf Reilmann MD</name><affiliations><json:string>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</json:string></affiliations></json:item><json:item><name>Stefan Bohlen MD</name><affiliations><json:string>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</json:string></affiliations></json:item><json:item><name>Thomas Klopstock MD</name><affiliations><json:string>Department of Neurology, Friedrich‐Baur‐Institute, Ludwig‐Maximilians‐University, Munich, Germany</json:string></affiliations></json:item><json:item><name>Andreas Bender MD</name><affiliations><json:string>Department of Neurology, Friedrich‐Baur‐Institute, Ludwig‐Maximilians‐University, Munich, Germany</json:string></affiliations></json:item><json:item><name>Adolf Weindl MD</name><affiliations><json:string>Department of Neurology, Technical University Munich, Munich, Germany</json:string></affiliations></json:item><json:item><name>Philipp Saemann MD</name><affiliations><json:string>Department of Radiology, Max Planck Institute of Psychiatry, Munich, Germany</json:string></affiliations></json:item><json:item><name>Dorothee P. Auer MD</name><affiliations><json:string>Department of Radiology, Max Planck Institute of Psychiatry, Munich, Germany</json:string></affiliations></json:item><json:item><name>E. Bernd Ringelstein MD</name><affiliations><json:string>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</json:string></affiliations></json:item><json:item><name>Herwig W. Lange MD</name><affiliations><json:string>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</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>chorea</value></json:item><json:item><lang><json:string>eng</json:string></lang><value>motor control</value></json:item><json:item><lang><json:string>eng</json:string></lang><value>basal ganglia</value></json:item></subject><language><json:string>eng</json:string></language><abstract>Motor symptoms in Huntington's Disease (HD) are commonly assessed by the Unified Huntington's Disease Rating Scale‐Total Motor Score (UHDRS‐TMS). However, the UHDRS‐TMS is limited by interrater variability, its categorical nature, and insensitivity in premanifest subjects. More objective and quantitative measures of motor phenotype may complement the use of the UHDRS‐TMS as outcome measure and increase the power and sensitivity of clinical trials. Deficits in tongue protrusion are well acknowledged in HD and constitute a subitem of the UHDRS‐TMS. We, therefore, investigated whether objective and quantitative assessment of tongue protrusion forces (TPF) provides measures that (1) correlate to the severity of motor phenotype detected in the UHDRS‐TMS in symptomatic HD, (2) detect a motor phenotype in premanifest HD gene‐carriers, and (3) exhibit a correlation to the genotype as assessed by a disease burden score (based on CAG‐repeat length and age). Using a precalibrated force transducer, the ability of premanifest gene carriers (n = 15) and subjects with symptomatic HD (n = 20) to generate and maintain isometric TPF at three target force levels (0.25, 0.5, and 1.0 N) was assessed and compared with age‐matched controls (n = 20) in a cross‐sectional study. Measures of variability of TPF and tongue contact time distinguished controls, premanifest, and symptomatic HD groups and correlated to the UHDRS‐TMS and disease burden score, suggesting a strong genotype‐phenotype correlation. Group distinction was most reliable at the lowest target force level. We conclude that assessment of TPF may be a useful objective and quantitative marker of motor dysfunction in premanifest and symptomatic HD. © 2010 Movement Disorder Society</abstract><qualityIndicators><score>6.848</score><pdfVersion>1.3</pdfVersion><pdfPageSize>612 x 810 pts</pdfPageSize><refBibsNative>true</refBibsNative><abstractCharCount>1745</abstractCharCount><pdfWordCount>3848</pdfWordCount><pdfCharCount>26437</pdfCharCount><pdfPageCount>8</pdfPageCount><abstractWordCount>259</abstractWordCount></qualityIndicators><title>Tongue force analysis assesses motor phenotype in premanifest and symptomatic Huntington's disease</title><genre><json:string>Serial article</json:string></genre><host><volume>25</volume><pages><total>8</total><last>2202</last><first>2195</first></pages><issn><json:string>0885-3185</json:string></issn><issue>13</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>2010</publicationDate><copyrightDate>2010</copyrightDate><doi><json:string>10.1002/mds.23243</json:string></doi><id>4340529D2182874F0A2B8EC8F45A449B0BDE6D06</id><fulltext><json:item><original>true</original><mimetype>application/pdf</mimetype><extension>pdf</extension><uri>https://api.istex.fr/document/4340529D2182874F0A2B8EC8F45A449B0BDE6D06/fulltext/pdf</uri></json:item><json:item><original>false</original><mimetype>application/zip</mimetype><extension>zip</extension><uri>https://api.istex.fr/document/4340529D2182874F0A2B8EC8F45A449B0BDE6D06/fulltext/zip</uri></json:item><istex:fulltextTEI uri="https://api.istex.fr/document/4340529D2182874F0A2B8EC8F45A449B0BDE6D06/fulltext/tei"><teiHeader type="text"><fileDesc><titleStmt><title level="a" type="main" xml:lang="en">Tongue force analysis assesses motor phenotype in premanifest and symptomatic Huntington's disease</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>2010</date></publicationStmt><notesStmt><note type="content">*Potential conflict of interest: The authors declare no financial interests related to this study.</note><note>Innovative Medical Research Fund (IMF)</note><note>Faculty of Medicine, University of Münster, Germany - No. RE‐120225;</note><note>Cure Huntington's Disease Initiative—Foundation (CHDI), New York, USA</note><note>European Huntington's Disease Network (EHDN—), Ulm, Germany [http://www.euro‐hd.net]</note></notesStmt><sourceDesc><biblStruct type="inbook"><analytic><title level="a" type="main" xml:lang="en">Tongue force analysis assesses motor phenotype in premanifest and symptomatic Huntington's disease</title><author><persName><forename type="first">Ralf</forename><surname>Reilmann</surname><roleName type="degree">MD</roleName></persName><note type="correspondence"><p>Correspondence: Department of Neurology, University Clinic Muenster (UKM), University of Muenster, Albert Schweitzer Strasse 33, 48129 Muenster, Germany</p></note><affiliation>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</affiliation></author><author><persName><forename type="first">Stefan</forename><surname>Bohlen</surname><roleName type="degree">MD</roleName></persName><affiliation>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</affiliation></author><author><persName><forename type="first">Thomas</forename><surname>Klopstock</surname><roleName type="degree">MD</roleName></persName><affiliation>Department of Neurology, Friedrich‐Baur‐Institute, Ludwig‐Maximilians‐University, Munich, Germany</affiliation></author><author><persName><forename type="first">Andreas</forename><surname>Bender</surname><roleName type="degree">MD</roleName></persName><affiliation>Department of Neurology, Friedrich‐Baur‐Institute, Ludwig‐Maximilians‐University, Munich, Germany</affiliation></author><author><persName><forename type="first">Adolf</forename><surname>Weindl</surname><roleName type="degree">MD</roleName></persName><affiliation>Department of Neurology, Technical University Munich, Munich, Germany</affiliation></author><author><persName><forename type="first">Philipp</forename><surname>Saemann</surname><roleName type="degree">MD</roleName></persName><affiliation>Department of Radiology, Max Planck Institute of Psychiatry, Munich, Germany</affiliation></author><author><persName><forename type="first">Dorothee P.</forename><surname>Auer</surname><roleName type="degree">MD</roleName></persName><affiliation>Department of Radiology, Max Planck Institute of Psychiatry, Munich, Germany</affiliation></author><author><persName><forename type="first">E. Bernd</forename><surname>Ringelstein</surname><roleName type="degree">MD</roleName></persName><affiliation>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</affiliation></author><author><persName><forename type="first">Herwig W.</forename><surname>Lange</surname><roleName type="degree">MD</roleName></persName><affiliation>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</affiliation></author></analytic><monogr><title level="j">Movement Disorders</title><title level="j" type="abbrev">Mov. 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However, the UHDRS‐TMS is limited by interrater variability, its categorical nature, and insensitivity in premanifest subjects. More objective and quantitative measures of motor phenotype may complement the use of the UHDRS‐TMS as outcome measure and increase the power and sensitivity of clinical trials. Deficits in tongue protrusion are well acknowledged in HD and constitute a subitem of the UHDRS‐TMS. We, therefore, investigated whether objective and quantitative assessment of tongue protrusion forces (TPF) provides measures that (1) correlate to the severity of motor phenotype detected in the UHDRS‐TMS in symptomatic HD, (2) detect a motor phenotype in premanifest HD gene‐carriers, and (3) exhibit a correlation to the genotype as assessed by a disease burden score (based on CAG‐repeat length and age). Using a precalibrated force transducer, the ability of premanifest gene carriers (n = 15) and subjects with symptomatic HD (n = 20) to generate and maintain isometric TPF at three target force levels (0.25, 0.5, and 1.0 N) was assessed and compared with age‐matched controls (n = 20) in a cross‐sectional study. Measures of variability of TPF and tongue contact time distinguished controls, premanifest, and symptomatic HD groups and correlated to the UHDRS‐TMS and disease burden score, suggesting a strong genotype‐phenotype correlation. Group distinction was most reliable at the lowest target force level. We conclude that assessment of TPF may be a useful objective and quantitative marker of motor dysfunction in premanifest and symptomatic HD. © 2010 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>chorea</term></item><item><term>motor control</term></item><item><term>basal ganglia</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="2009-07-15">Received</change><change when="2010-04-13">Registration</change><change when="2010-10-15">Published</change></revisionDesc></teiHeader></istex:fulltextTEI><json:item><original>false</original><mimetype>text/plain</mimetype><extension>txt</extension><uri>https://api.istex.fr/document/4340529D2182874F0A2B8EC8F45A449B0BDE6D06/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. Disord.</title></titleGroup></publicationMeta><publicationMeta level="part" position="130"><doi origin="wiley" registered="yes">10.1002/mds.v25:13</doi><numberingGroup><numbering type="journalVolume" number="25">25</numbering><numbering type="journalIssue">13</numbering></numberingGroup><coverDate startDate="2010-10-15">15 October 2010</coverDate></publicationMeta><publicationMeta level="unit" type="article" position="270" status="forIssue"><doi origin="wiley" registered="yes">10.1002/mds.23243</doi><idGroup><id type="unit" value="MDS23243"></id></idGroup><countGroup><count type="pageTotal" number="8"></count></countGroup><titleGroup><title type="articleCategory">Research Article</title><title type="tocHeading1">Research Articles</title></titleGroup><copyright ownership="thirdParty">Copyright © 2010 Movement Disorder Society</copyright><eventGroup><event type="manuscriptReceived" date="2009-07-15"></event><event type="manuscriptRevised" date="2010-03-15"></event><event type="manuscriptAccepted" date="2010-04-13"></event><event type="firstOnline" date="2010-07-19"></event><event type="publishedOnlineAcceptedOrEarlyUnpaginated" date="2010-07-19"></event><event type="xmlConverted" agent="Converter:JWSART34_TO_WML3G version:2.5.2 mode:FullText source:FullText result:FullText" date="2011-07-06"></event><event type="publishedOnlineFinalForm" date="2010-07-19"></event><event type="xmlConverted" agent="Converter:WILEY_ML3G_TO_WILEY_ML3GV2 version:3.8.8" date="2014-02-02"></event><event type="xmlConverted" agent="Converter:WML3G_To_WML3G version:4.1.7 mode:FullText,remove_FC" date="2014-10-31"></event></eventGroup><numberingGroup><numbering type="pageFirst">2195</numbering><numbering type="pageLast">2202</numbering></numberingGroup><correspondenceTo>Department of Neurology, University Clinic Muenster (UKM), University of Muenster, Albert Schweitzer Strasse 33, 48129 Muenster, Germany</correspondenceTo><linkGroup><link type="toTypesetVersion" href="file:MDS.MDS23243.pdf"></link></linkGroup></publicationMeta><contentMeta><countGroup><count type="figureTotal" number="3"></count><count type="tableTotal" number="2"></count><count type="referenceTotal" number="21"></count><count type="wordTotal" number="6332"></count></countGroup><titleGroup><title type="main" xml:lang="en">Tongue force analysis assesses motor phenotype in premanifest and symptomatic Huntington's disease<link href="#fn7"></link></title><title type="short" xml:lang="en">Motor Phenotype Assessment in HD</title></titleGroup><creators><creator xml:id="au1" creatorRole="author" affiliationRef="#af1" corresponding="yes"><personName><givenNames>Ralf</givenNames><familyName>Reilmann</familyName><degrees>MD</degrees></personName><contactDetails><email normalForm="r.reilmann@uni-muenster.de">r.reilmann@uni‐muenster.de</email></contactDetails></creator><creator xml:id="au2" creatorRole="author" affiliationRef="#af1"><personName><givenNames>Stefan</givenNames><familyName>Bohlen</familyName><degrees>MD</degrees></personName></creator><creator xml:id="au3" creatorRole="author" affiliationRef="#af2"><personName><givenNames>Thomas</givenNames><familyName>Klopstock</familyName><degrees>MD</degrees></personName></creator><creator xml:id="au4" creatorRole="author" affiliationRef="#af2"><personName><givenNames>Andreas</givenNames><familyName>Bender</familyName><degrees>MD</degrees></personName></creator><creator xml:id="au5" creatorRole="author" affiliationRef="#af3"><personName><givenNames>Adolf</givenNames><familyName>Weindl</familyName><degrees>MD</degrees></personName></creator><creator xml:id="au6" creatorRole="author" affiliationRef="#af4"><personName><givenNames>Philipp</givenNames><familyName>Saemann</familyName><degrees>MD</degrees></personName></creator><creator xml:id="au7" creatorRole="author" affiliationRef="#af4"><personName><givenNames>Dorothee P.</givenNames><familyName>Auer</familyName><degrees>MD</degrees></personName></creator><creator xml:id="au8" creatorRole="author" affiliationRef="#af1"><personName><givenNames>E. Bernd</givenNames><familyName>Ringelstein</familyName><degrees>MD</degrees></personName></creator><creator xml:id="au9" creatorRole="author" affiliationRef="#af1"><personName><givenNames>Herwig W.</givenNames><familyName>Lange</familyName><degrees>MD</degrees></personName></creator></creators><affiliationGroup><affiliation xml:id="af1" countryCode="DE" type="organization"><unparsedAffiliation>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</unparsedAffiliation></affiliation><affiliation xml:id="af2" countryCode="DE" type="organization"><unparsedAffiliation>Department of Neurology, Friedrich‐Baur‐Institute, Ludwig‐Maximilians‐University, Munich, Germany</unparsedAffiliation></affiliation><affiliation xml:id="af3" countryCode="DE" type="organization"><unparsedAffiliation>Department of Neurology, Technical University Munich, Munich, Germany</unparsedAffiliation></affiliation><affiliation xml:id="af4" countryCode="DE" type="organization"><unparsedAffiliation>Department of Radiology, Max Planck Institute of Psychiatry, Munich, Germany</unparsedAffiliation></affiliation></affiliationGroup><keywordGroup xml:lang="en" type="author"><keyword xml:id="kwd1">Huntington's disease</keyword><keyword xml:id="kwd2">chorea</keyword><keyword xml:id="kwd3">motor control</keyword><keyword xml:id="kwd4">basal ganglia</keyword></keywordGroup><fundingInfo><fundingAgency>Innovative Medical Research Fund (IMF)</fundingAgency></fundingInfo><fundingInfo><fundingAgency>Faculty of Medicine, University of Münster, Germany</fundingAgency><fundingNumber>RE‐120225</fundingNumber></fundingInfo><fundingInfo><fundingAgency>Cure Huntington's Disease Initiative—Foundation (CHDI), New York, USA</fundingAgency></fundingInfo><fundingInfo><fundingAgency>European Huntington's Disease Network (EHDN—), Ulm, Germany [http://www.euro‐hd.net]</fundingAgency></fundingInfo><abstractGroup><abstract type="main" xml:lang="en"><title type="main">Abstract</title><p>Motor symptoms in Huntington's Disease (HD) are commonly assessed by the Unified Huntington's Disease Rating Scale‐Total Motor Score (UHDRS‐TMS). However, the UHDRS‐TMS is limited by interrater variability, its categorical nature, and insensitivity in premanifest subjects. More objective and quantitative measures of motor phenotype may complement the use of the UHDRS‐TMS as outcome measure and increase the power and sensitivity of clinical trials. Deficits in tongue protrusion are well acknowledged in HD and constitute a subitem of the UHDRS‐TMS. We, therefore, investigated whether objective and quantitative assessment of tongue protrusion forces (TPF) provides measures that (1) correlate to the severity of motor phenotype detected in the UHDRS‐TMS in symptomatic HD, (2) detect a motor phenotype in premanifest HD gene‐carriers, and (3) exhibit a correlation to the genotype as assessed by a disease burden score (based on CAG‐repeat length and age). Using a precalibrated force transducer, the ability of premanifest gene carriers (n = 15) and subjects with symptomatic HD (n = 20) to generate and maintain isometric TPF at three target force levels (0.25, 0.5, and 1.0 N) was assessed and compared with age‐matched controls (n = 20) in a cross‐sectional study. Measures of variability of TPF and tongue contact time distinguished controls, premanifest, and symptomatic HD groups and correlated to the UHDRS‐TMS and disease burden score, suggesting a strong genotype‐phenotype correlation. Group distinction was most reliable at the lowest target force level. We conclude that assessment of TPF may be a useful objective and quantitative marker of motor dysfunction in premanifest and symptomatic HD. © 2010 Movement Disorder Society</p></abstract></abstractGroup></contentMeta><noteGroup><note xml:id="fn7"><p>Potential conflict of interest: The authors declare no financial interests related to this study.</p></note></noteGroup></header></component></istex:document></istex:metadataXml><!--Version 0.6 générée le 3-12-2015--><mods version="3.6"><titleInfo lang="en"><title>Tongue force analysis assesses motor phenotype in premanifest and symptomatic Huntington's disease</title></titleInfo><titleInfo type="abbreviated" lang="en"><title>Motor Phenotype Assessment in HD</title></titleInfo><titleInfo type="alternative" contentType="CDATA" lang="en"><title>Tongue force analysis assesses motor phenotype in premanifest and symptomatic Huntington's disease</title></titleInfo><name type="personal"><namePart type="given">Ralf</namePart><namePart type="family">Reilmann</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</affiliation><description>Correspondence: Department of Neurology, University Clinic Muenster (UKM), University of Muenster, Albert Schweitzer Strasse 33, 48129 Muenster, Germany</description><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Stefan</namePart><namePart type="family">Bohlen</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Thomas</namePart><namePart type="family">Klopstock</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Neurology, Friedrich‐Baur‐Institute, Ludwig‐Maximilians‐University, Munich, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Andreas</namePart><namePart type="family">Bender</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Neurology, Friedrich‐Baur‐Institute, Ludwig‐Maximilians‐University, Munich, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Adolf</namePart><namePart type="family">Weindl</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Neurology, Technical University Munich, Munich, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Philipp</namePart><namePart type="family">Saemann</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Radiology, Max Planck Institute of Psychiatry, Munich, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Dorothee P.</namePart><namePart type="family">Auer</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Radiology, Max Planck Institute of Psychiatry, Munich, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">E. Bernd</namePart><namePart type="family">Ringelstein</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</affiliation><role><roleTerm type="text">author</roleTerm></role></name><name type="personal"><namePart type="given">Herwig W.</namePart><namePart type="family">Lange</namePart><namePart type="termsOfAddress">MD</namePart><affiliation>Department of Neurology, University Clinic Muenster (UKM), Westfaelische Wilhelms University of Muenster, Muenster, Germany</affiliation><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">2010-10-15</dateIssued><dateCaptured encoding="w3cdtf">2009-07-15</dateCaptured><dateValid encoding="w3cdtf">2010-04-13</dateValid><copyrightDate encoding="w3cdtf">2010</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">3</extent><extent unit="tables">2</extent><extent unit="references">21</extent><extent unit="words">6332</extent></physicalDescription><abstract lang="en">Motor symptoms in Huntington's Disease (HD) are commonly assessed by the Unified Huntington's Disease Rating Scale‐Total Motor Score (UHDRS‐TMS). However, the UHDRS‐TMS is limited by interrater variability, its categorical nature, and insensitivity in premanifest subjects. More objective and quantitative measures of motor phenotype may complement the use of the UHDRS‐TMS as outcome measure and increase the power and sensitivity of clinical trials. Deficits in tongue protrusion are well acknowledged in HD and constitute a subitem of the UHDRS‐TMS. We, therefore, investigated whether objective and quantitative assessment of tongue protrusion forces (TPF) provides measures that (1) correlate to the severity of motor phenotype detected in the UHDRS‐TMS in symptomatic HD, (2) detect a motor phenotype in premanifest HD gene‐carriers, and (3) exhibit a correlation to the genotype as assessed by a disease burden score (based on CAG‐repeat length and age). Using a precalibrated force transducer, the ability of premanifest gene carriers (n = 15) and subjects with symptomatic HD (n = 20) to generate and maintain isometric TPF at three target force levels (0.25, 0.5, and 1.0 N) was assessed and compared with age‐matched controls (n = 20) in a cross‐sectional study. Measures of variability of TPF and tongue contact time distinguished controls, premanifest, and symptomatic HD groups and correlated to the UHDRS‐TMS and disease burden score, suggesting a strong genotype‐phenotype correlation. Group distinction was most reliable at the lowest target force level. We conclude that assessment of TPF may be a useful objective and quantitative marker of motor dysfunction in premanifest and symptomatic HD. © 2010 Movement Disorder Society</abstract><note type="content">*Potential conflict of interest: The authors declare no financial interests related to this study.</note><note type="funding">Innovative Medical Research Fund (IMF)</note><note type="funding">Faculty of Medicine, University of Münster, Germany - No. RE‐120225; </note><note type="funding">Cure Huntington's Disease Initiative—Foundation (CHDI), New York, USA</note><note type="funding">European Huntington's Disease Network (EHDN—), Ulm, Germany [http://www.euro‐hd.net]</note><subject lang="en"><genre>Keywords</genre><topic>Huntington's disease</topic><topic>chorea</topic><topic>motor control</topic><topic>basal ganglia</topic></subject><relatedItem type="host"><titleInfo><title>Movement Disorders</title></titleInfo><titleInfo type="abbreviated"><title>Mov. 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