Motor abnormalities in premanifest persons with Huntington's disease: The PREDICT‐HD study
Identifieur interne : 002209 ( Main/Exploration ); précédent : 002208; suivant : 002210Motor 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.
Descripteurs français
- Pascal (Inist)
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 disease, Huntington's disease, Magnetic Resonance Imaging (methods), Male, Middle Aged, Motor Activity (genetics), Motor Activity (physiology), Nervous system diseases, Neuropsychological Tests, Probability, Regression Analysis, Retrospective Studies, Risk factor, 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
Affiliations:
- Australie, États-Unis
- Indiana, Iowa, Maryland, New Jersey, Washington (État), État de New York
- Iowa City, Seattle
- Université de Washington, Université de l'Iowa
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Le document en format XML
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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 disease</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>Nervous system diseases</term><term>Neuropsychological Tests</term><term>Probability</term><term>Regression Analysis</term><term>Retrospective Studies</term><term>Risk factor</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><keywords scheme="Pascal" xml:lang="fr"><term>Chorée de Huntington</term><term>Facteur risque</term><term>Pathologie du système nerveux</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><affiliations><list><country><li>Australie</li><li>États-Unis</li></country><region><li>Indiana</li><li>Iowa</li><li>Maryland</li><li>New Jersey</li><li>Washington (État)</li><li>État de New York</li></region><settlement><li>Iowa City</li><li>Seattle</li></settlement><orgName><li>Université de Washington</li><li>Université de l'Iowa</li></orgName></list><tree><country name="États-Unis"><region name="État de New York"><name sortKey="Biglan, Kevin M" sort="Biglan, Kevin M" uniqKey="Biglan K" first="Kevin M." last="Biglan">Kevin M. Biglan</name></region><name sortKey="Aylward, Elizabeth H" sort="Aylward, Elizabeth H" uniqKey="Aylward E" first="Elizabeth H." last="Aylward">Elizabeth H. Aylward</name><name sortKey="Beglinger, Leigh J" sort="Beglinger, Leigh J" uniqKey="Beglinger L" first="Leigh J." last="Beglinger">Leigh J. Beglinger</name><name sortKey="Carlozzi, Noelle E" sort="Carlozzi, Noelle E" uniqKey="Carlozzi N" first="Noelle E." last="Carlozzi">Noelle E. Carlozzi</name><name sortKey="Duff, Kevin" sort="Duff, Kevin" uniqKey="Duff K" first="Kevin" last="Duff">Kevin Duff</name><name sortKey="Langbehn, Douglas R" sort="Langbehn, Douglas R" uniqKey="Langbehn D" first="Douglas R." last="Langbehn">Douglas R. Langbehn</name><name sortKey="Paulsen, Jane S" sort="Paulsen, Jane S" uniqKey="Paulsen J" first="Jane S." last="Paulsen">Jane S. Paulsen</name><name sortKey="Queller, Sarah" sort="Queller, Sarah" uniqKey="Queller S" first="Sarah" last="Queller">Sarah Queller</name><name sortKey="Ross, Christopher A" sort="Ross, Christopher A" uniqKey="Ross C" first="Christopher A." last="Ross">Christopher A. Ross</name></country><country name="Australie"><noRegion><name sortKey="Stout, Julie C" sort="Stout, Julie C" uniqKey="Stout J" first="Julie C." last="Stout">Julie C. Stout</name></noRegion></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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