Motor abnormalities in premanifest persons with Huntington's disease: The PREDICT‐HD study
Identifieur interne : 002A70 ( Main/Merge ); précédent : 002A69; suivant : 002A71Motor abnormalities in premanifest persons with Huntington's disease: The PREDICT‐HD study
Auteurs : Kevin M. Biglan [États-Unis] ; Christopher A. Ross [États-Unis] ; Douglas R. Langbehn [États-Unis] ; Elizabeth H. Aylward [États-Unis] ; Julie C. Stout [Australie] ; Sarah Queller [États-Unis] ; Noelle E. Carlozzi [États-Unis] ; Kevin Duff [États-Unis] ; Leigh J. Beglinger [États-Unis] ; Jane S. Paulsen [États-Unis]Source :
- Movement Disorders [ 0885-3185 ] ; 2009-09-15.
English descriptors
- KwdEn :
- Adult, Chi-Square Distribution, Corpus Striatum (pathology), Female, Genetic Testing (methods), Humans, Huntington Disease (diagnosis), Huntington Disease (genetics), Huntington Disease (physiopathology), Huntington's disease, Magnetic Resonance Imaging (methods), Male, Middle Aged, Motor Activity (genetics), Motor Activity (physiology), Neuropsychological Tests, Probability, Regression Analysis, Retrospective Studies, Trinucleotide Repeats (genetics), UHDRS, at‐risk.
- MESH :
- diagnosis : Huntington Disease.
- genetics : Huntington Disease, Motor Activity, Trinucleotide Repeats.
- methods : Genetic Testing, Magnetic Resonance Imaging.
- pathology : Corpus Striatum.
- physiology : Motor Activity.
- physiopathology : Huntington Disease.
- Adult, Chi-Square Distribution, Female, Humans, Male, Middle Aged, Neuropsychological Tests, Probability, Regression Analysis, Retrospective Studies.
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
Url:
- https://api.istex.fr/document/6AA2C4AC0C2B0FC427531284AAF17804A28580B2/fulltext/pdf
- http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3048804
DOI: 10.1002/mds.22601
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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>Adult</term><term>Chi-Square Distribution</term><term>Corpus Striatum (pathology)</term><term>Female</term><term>Genetic Testing (methods)</term><term>Humans</term><term>Huntington Disease (diagnosis)</term><term>Huntington Disease (genetics)</term><term>Huntington Disease (physiopathology)</term><term>Huntington's disease</term><term>Magnetic Resonance Imaging (methods)</term><term>Male</term><term>Middle Aged</term><term>Motor Activity (genetics)</term><term>Motor Activity (physiology)</term><term>Neuropsychological Tests</term><term>Probability</term><term>Regression Analysis</term><term>Retrospective Studies</term><term>Trinucleotide Repeats (genetics)</term><term>UHDRS</term><term>at‐risk</term></keywords><keywords scheme="MESH" qualifier="diagnosis" xml:lang="en"><term>Huntington Disease</term></keywords><keywords scheme="MESH" qualifier="genetics" xml:lang="en"><term>Huntington Disease</term><term>Motor Activity</term><term>Trinucleotide Repeats</term></keywords><keywords scheme="MESH" qualifier="methods" xml:lang="en"><term>Genetic Testing</term><term>Magnetic Resonance Imaging</term></keywords><keywords scheme="MESH" qualifier="pathology" xml:lang="en"><term>Corpus Striatum</term></keywords><keywords scheme="MESH" qualifier="physiology" xml:lang="en"><term>Motor Activity</term></keywords><keywords scheme="MESH" qualifier="physiopathology" xml:lang="en"><term>Huntington Disease</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adult</term><term>Chi-Square Distribution</term><term>Female</term><term>Humans</term><term>Male</term><term>Middle Aged</term><term>Neuropsychological Tests</term><term>Probability</term><term>Regression Analysis</term><term>Retrospective Studies</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><double doi="10.1002/mds.22601"><ISTEX><TEI wicri:istexFullTextTei="biblStruct"><teiHeader><fileDesc><titleStmt><title xml:lang="en">Motor abnormalities in premanifest persons with Huntington's disease: The PREDICT‐HD study</title><author><name sortKey="Biglan, Kevin M" sort="Biglan, Kevin M" uniqKey="Biglan K" first="Kevin M." last="Biglan">Kevin M. Biglan</name></author><author><name sortKey="Ross, Christopher A" sort="Ross, Christopher A" uniqKey="Ross C" first="Christopher A." last="Ross">Christopher A. Ross</name></author><author><name sortKey="Langbehn, Douglas R" sort="Langbehn, Douglas R" uniqKey="Langbehn D" first="Douglas R." last="Langbehn">Douglas R. Langbehn</name></author><author><name sortKey="Aylward, Elizabeth H" sort="Aylward, Elizabeth H" uniqKey="Aylward E" first="Elizabeth H." last="Aylward">Elizabeth H. Aylward</name></author><author><name sortKey="Stout, Julie C" sort="Stout, Julie C" uniqKey="Stout J" first="Julie C." last="Stout">Julie C. Stout</name></author><author><name sortKey="Queller, Sarah" sort="Queller, Sarah" uniqKey="Queller S" first="Sarah" last="Queller">Sarah Queller</name></author><author><name sortKey="Carlozzi, Noelle E" sort="Carlozzi, Noelle E" uniqKey="Carlozzi N" first="Noelle E." last="Carlozzi">Noelle E. Carlozzi</name></author><author><name sortKey="Duff, Kevin" sort="Duff, Kevin" uniqKey="Duff K" first="Kevin" last="Duff">Kevin Duff</name></author><author><name sortKey="Beglinger, Leigh J" sort="Beglinger, Leigh J" uniqKey="Beglinger L" first="Leigh J." last="Beglinger">Leigh J. Beglinger</name></author><author><name sortKey="Paulsen, Jane S" sort="Paulsen, Jane S" uniqKey="Paulsen J" first="Jane S." last="Paulsen">Jane S. Paulsen</name></author></titleStmt><publicationStmt><idno type="wicri:source">ISTEX</idno><idno type="RBID">ISTEX:6AA2C4AC0C2B0FC427531284AAF17804A28580B2</idno><date when="2009" year="2009">2009</date><idno type="doi">10.1002/mds.22601</idno><idno type="url">https://api.istex.fr/document/6AA2C4AC0C2B0FC427531284AAF17804A28580B2/fulltext/pdf</idno><idno type="wicri:Area/Istex/Corpus">000561</idno><idno type="wicri:Area/Istex/Curation">000561</idno><idno type="wicri:Area/Istex/Checkpoint">000E11</idno><idno type="wicri:doubleKey">0885-3185:2009:Biglan K:motor:abnormalities:in</idno></publicationStmt><sourceDesc><biblStruct><analytic><title level="a" type="main" xml:lang="en">Motor abnormalities in premanifest persons with Huntington's disease: The PREDICT‐HD study</title><author><name sortKey="Biglan, Kevin M" sort="Biglan, Kevin M" uniqKey="Biglan K" first="Kevin M." last="Biglan">Kevin M. Biglan</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>University of Rochester, Rochester, New York</wicri:regionArea><placeName><region type="state">État de New York</region></placeName></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 wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Johns Hopkins University, Baltimore, Maryland</wicri:regionArea><placeName><region type="state">Maryland</region></placeName></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 wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>University of Iowa, Iowa City, Iowa</wicri:regionArea><placeName><region type="state">Iowa</region><settlement type="city">Iowa City</settlement></placeName><orgName type="university">Université de l'Iowa</orgName></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 wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>University of Washington, Seattle, Washington</wicri:regionArea><placeName><region type="state">Washington (État)</region><settlement type="city">Seattle</settlement></placeName><orgName type="university">Université de Washington</orgName></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 wicri:level="1"><country xml:lang="fr">Australie</country><wicri:regionArea>Monash University, Victoria</wicri:regionArea><wicri:noRegion>Victoria</wicri:noRegion></affiliation></author><author><name sortKey="Queller, Sarah" sort="Queller, Sarah" uniqKey="Queller S" first="Sarah" last="Queller">Sarah Queller</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Indiana University, Bloomington, Indiana</wicri:regionArea><placeName><region type="state">Indiana</region></placeName></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 wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Kessler Medical Rehabilitation Research and Education Center, West Orange, New Jersey</wicri:regionArea><placeName><region type="state">New Jersey</region></placeName></affiliation></author><author><name sortKey="Duff, Kevin" sort="Duff, Kevin" uniqKey="Duff K" first="Kevin" last="Duff">Kevin Duff</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>University of Iowa, Iowa City, Iowa</wicri:regionArea><placeName><region type="state">Iowa</region><settlement type="city">Iowa City</settlement></placeName><orgName type="university">Université de l'Iowa</orgName></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 wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>University of Iowa, Iowa City, Iowa</wicri:regionArea><placeName><region type="state">Iowa</region><settlement type="city">Iowa City</settlement></placeName><orgName type="university">Université de l'Iowa</orgName></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 wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>University of Iowa, Iowa City, Iowa</wicri:regionArea><placeName><region type="state">Iowa</region><settlement type="city">Iowa City</settlement></placeName><orgName type="university">Université de l'Iowa</orgName></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><PMC><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Motor Abnormalities in Premanifest Persons with Huntington’s Disease: The PREDICT-HD Study</title><author><name sortKey="Biglan, Kevin M" sort="Biglan, Kevin M" uniqKey="Biglan K" first="Kevin M." last="Biglan">Kevin M. Biglan</name><affiliation wicri:level="2"><nlm:aff id="A1"> University of Rochester, Rochester, New York</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">État de New York</region></placeName><wicri:cityArea> University of Rochester, Rochester</wicri:cityArea></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 wicri:level="2"><nlm:aff id="A2"> Johns Hopkins University, Baltimore, Maryland</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Maryland</region></placeName><wicri:cityArea> Johns Hopkins University, Baltimore</wicri:cityArea></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 wicri:level="2"><nlm:aff id="A3"> University of Iowa, Iowa City, Iowa</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Iowa</region></placeName><wicri:cityArea> University of Iowa, Iowa City</wicri:cityArea></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 wicri:level="2"><nlm:aff id="A4"> University of Washington, Seattle, Washington</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Washington (État)</region></placeName><wicri:cityArea> University of Washington, Seattle</wicri:cityArea></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 wicri:level="1"><nlm:aff id="A5"> Monash University, Victoria, Australia</nlm:aff><country xml:lang="fr">Australie</country><wicri:regionArea> Monash University, Victoria</wicri:regionArea><wicri:noRegion>Victoria</wicri:noRegion></affiliation></author><author><name sortKey="Queller, Sarah" sort="Queller, Sarah" uniqKey="Queller S" first="Sarah" last="Queller">Sarah Queller</name><affiliation wicri:level="2"><nlm:aff id="A6"> Indiana University, Bloomington, Indiana</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Indiana</region></placeName><wicri:cityArea> Indiana University, Bloomington</wicri:cityArea></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 wicri:level="2"><nlm:aff id="A7"> Kessler Medical Rehabilitation Research & Education Center, West Orange, New Jersey</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">New Jersey</region></placeName><wicri:cityArea> Kessler Medical Rehabilitation Research & Education Center, West Orange</wicri:cityArea></affiliation></author><author><name sortKey="Duff, Kevin" sort="Duff, Kevin" uniqKey="Duff K" first="Kevin" last="Duff">Kevin Duff</name><affiliation wicri:level="2"><nlm:aff id="A3"> University of Iowa, Iowa City, Iowa</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Iowa</region></placeName><wicri:cityArea> University of Iowa, Iowa City</wicri:cityArea></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 wicri:level="2"><nlm:aff id="A3"> University of Iowa, Iowa City, Iowa</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Iowa</region></placeName><wicri:cityArea> University of Iowa, Iowa City</wicri:cityArea></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 wicri:level="2"><nlm:aff id="A3"> University of Iowa, Iowa City, Iowa</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Iowa</region></placeName><wicri:cityArea> University of Iowa, Iowa City</wicri:cityArea></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">19562761</idno><idno type="pmc">3048804</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3048804</idno><idno type="RBID">PMC:3048804</idno><idno type="doi">10.1002/mds.22601</idno><date when="2009">2009</date><idno type="wicri:Area/Pmc/Corpus">000065</idno><idno type="wicri:Area/Pmc/Curation">000065</idno><idno type="wicri:Area/Pmc/Checkpoint">000402</idno><idno type="wicri:source">PubMed</idno><idno type="wicri:Area/PubMed/Corpus">001C41</idno><idno type="wicri:Area/PubMed/Curation">001C41</idno><idno type="wicri:Area/PubMed/Checkpoint">001D16</idno><idno type="wicri:Area/Ncbi/Merge">002736</idno><idno type="wicri:Area/Ncbi/Curation">002736</idno><idno type="wicri:Area/Ncbi/Checkpoint">002736</idno><idno type="wicri:doubleKey">0885-3185:2009:Biglan K:motor:abnormalities:in</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">Motor Abnormalities in Premanifest Persons with Huntington’s Disease: The PREDICT-HD Study</title><author><name sortKey="Biglan, Kevin M" sort="Biglan, Kevin M" uniqKey="Biglan K" first="Kevin M." last="Biglan">Kevin M. Biglan</name><affiliation wicri:level="2"><nlm:aff id="A1"> University of Rochester, Rochester, New York</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">État de New York</region></placeName><wicri:cityArea> University of Rochester, Rochester</wicri:cityArea></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 wicri:level="2"><nlm:aff id="A2"> Johns Hopkins University, Baltimore, Maryland</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Maryland</region></placeName><wicri:cityArea> Johns Hopkins University, Baltimore</wicri:cityArea></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 wicri:level="2"><nlm:aff id="A3"> University of Iowa, Iowa City, Iowa</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Iowa</region></placeName><wicri:cityArea> University of Iowa, Iowa City</wicri:cityArea></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 wicri:level="2"><nlm:aff id="A4"> University of Washington, Seattle, Washington</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Washington (État)</region></placeName><wicri:cityArea> University of Washington, Seattle</wicri:cityArea></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 wicri:level="1"><nlm:aff id="A5"> Monash University, Victoria, Australia</nlm:aff><country xml:lang="fr">Australie</country><wicri:regionArea> Monash University, Victoria</wicri:regionArea><wicri:noRegion>Victoria</wicri:noRegion></affiliation></author><author><name sortKey="Queller, Sarah" sort="Queller, Sarah" uniqKey="Queller S" first="Sarah" last="Queller">Sarah Queller</name><affiliation wicri:level="2"><nlm:aff id="A6"> Indiana University, Bloomington, Indiana</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Indiana</region></placeName><wicri:cityArea> Indiana University, Bloomington</wicri:cityArea></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 wicri:level="2"><nlm:aff id="A7"> Kessler Medical Rehabilitation Research & Education Center, West Orange, New Jersey</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">New Jersey</region></placeName><wicri:cityArea> Kessler Medical Rehabilitation Research & Education Center, West Orange</wicri:cityArea></affiliation></author><author><name sortKey="Duff, Kevin" sort="Duff, Kevin" uniqKey="Duff K" first="Kevin" last="Duff">Kevin Duff</name><affiliation wicri:level="2"><nlm:aff id="A3"> University of Iowa, Iowa City, Iowa</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Iowa</region></placeName><wicri:cityArea> University of Iowa, Iowa City</wicri:cityArea></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 wicri:level="2"><nlm:aff id="A3"> University of Iowa, Iowa City, Iowa</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Iowa</region></placeName><wicri:cityArea> University of Iowa, Iowa City</wicri:cityArea></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 wicri:level="2"><nlm:aff id="A3"> University of Iowa, Iowa City, Iowa</nlm:aff><country xml:lang="fr">États-Unis</country><placeName><region type="state">Iowa</region></placeName><wicri:cityArea> University of Iowa, Iowa City</wicri:cityArea></affiliation></author></analytic><series><title level="j">Movement disorders : official journal of the Movement Disorder Society</title><idno type="ISSN">0885-3185</idno><idno type="eISSN">1531-8257</idno><imprint><date when="2009">2009</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adult</term><term>Chi-Square Distribution</term><term>Corpus Striatum (pathology)</term><term>Female</term><term>Genetic Testing (methods)</term><term>Humans</term><term>Huntington Disease (diagnosis)</term><term>Huntington Disease (genetics)</term><term>Huntington Disease (physiopathology)</term><term>Magnetic Resonance Imaging (methods)</term><term>Male</term><term>Middle Aged</term><term>Motor Activity (genetics)</term><term>Motor Activity (physiology)</term><term>Neuropsychological Tests</term><term>Probability</term><term>Regression Analysis</term><term>Retrospective Studies</term><term>Trinucleotide Repeats (genetics)</term></keywords><keywords scheme="MESH" qualifier="diagnosis" xml:lang="en"><term>Huntington Disease</term></keywords><keywords scheme="MESH" qualifier="genetics" xml:lang="en"><term>Huntington Disease</term><term>Motor Activity</term><term>Trinucleotide Repeats</term></keywords><keywords scheme="MESH" qualifier="methods" xml:lang="en"><term>Genetic Testing</term><term>Magnetic Resonance Imaging</term></keywords><keywords scheme="MESH" qualifier="pathology" xml:lang="en"><term>Corpus Striatum</term></keywords><keywords scheme="MESH" qualifier="physiology" xml:lang="en"><term>Motor Activity</term></keywords><keywords scheme="MESH" qualifier="physiopathology" xml:lang="en"><term>Huntington Disease</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adult</term><term>Chi-Square Distribution</term><term>Female</term><term>Humans</term><term>Male</term><term>Middle Aged</term><term>Neuropsychological Tests</term><term>Probability</term><term>Regression Analysis</term><term>Retrospective Studies</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><sec id="S1"><title>Background</title><p id="P1">The PREDICT-HD study seeks to identify clinical and biological markers of Huntington’s disease in premanifest individuals who have undergone predictive genetic testing.</p></sec><sec sec-type="methods" id="S2"><title>Methods</title><p id="P2">We compared baseline motor data between gene-expansion carriers (cases) and non gene-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 MRI measurements. Multiple linear regression associated total motor score, motor domains and individual motor items with estimated diagnosis and striatal volumes.</p></sec><sec id="S3"><title>Results</title><p id="P3">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.</p></sec><sec id="S4"><title>Conclusions</title><p id="P4">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.</p></sec></div></front></TEI></PMC></double></record>Pour manipuler ce document sous Unix (Dilib)
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