An Item Response Analysis of the Motor and Behavioral Subscales of the Unified Huntington's Disease Rating Scale in Huntington Disease Gene Expansion Carriers
Identifieur interne : 003009 ( Ncbi/Curation ); précédent : 003008; suivant : 003010An Item Response Analysis of the Motor and Behavioral Subscales of the Unified Huntington's Disease Rating Scale in Huntington Disease Gene Expansion Carriers
Auteurs : Anthony L. Vaccarino [Canada] ; Karen Anderson [États-Unis] ; Beth Borowsky [États-Unis] ; Kevin Duff [États-Unis] ; Joseph Giuliano [États-Unis] ; Mark Guttman [Canada] ; Aileen K. Ho [Royaume-Uni] ; Michael Orth [Allemagne] ; Jane S. Paulsen [États-Unis] ; Terrence Sills [Canada] ; Daniel P. Van Kammen [États-Unis] ; Kenneth R. Evans [Canada]Source :
- Movement disorders : official journal of the Movement Disorder Society [ 0885-3185 ] ; 2011.
English descriptors
- KwdEn :
- Adolescent, Adult, Aged, Aged, 80 and over, Behavioral Symptoms (etiology), Disability Evaluation, Female, Humans, Huntington Disease (complications), Huntington Disease (diagnosis), Huntington Disease (genetics), Male, Middle Aged, Movement Disorders (etiology), Severity of Illness Index, Trinucleotide Repeat Expansion (genetics), Young Adult.
- MESH :
- complications : Huntington Disease.
- diagnosis : Huntington Disease.
- etiology : Behavioral Symptoms, Movement Disorders.
- genetics : Huntington Disease, Trinucleotide Repeat Expansion.
- Adolescent, Adult, Aged, Aged, 80 and over, Disability Evaluation, Female, Humans, Male, Middle Aged, Severity of Illness Index, Young Adult.
Abstract
Although the Unified Huntington's Disease Rating Scale (UHDRS) is widely used in the assessment of Huntington disease (HD), the ability of individual items to discriminate individual differences in motor or behavioral manifestations has not been extensively studied in HD gene expansion carriers without a motor-defined clinical diagnosis (i.e., prodromal-HD or prHD). To elucidate the relationship between scores on individual motor and behavioral UHDRS items and total score for each subscale, a non-parametric item response analysis was performed on retrospective data from two multicentre, longitudinal studies. Motor and Behavioral assessments were supplied for 737 prHD individuals with data from 2114 visits (PREDICT-HD) and 686 HD individuals with data from 1482 visits (REGISTRY). Option characteristic curves were generated for UHDRS subscale items in relation to their subscale score. In prHD, overall severity of motor signs was low and participants had scores of 2 or above on very few items. In HD, motor items that assessed ocular pursuit, saccade initiation, finger tapping, tandem walking, and to a lesser extent saccade velocity, dysarthia, tongue protrusion, pronation/supination, Luria, bradykinesia, choreas, gait and balance on the retropulsion test were found to discriminate individual differences across a broad range of motor severity. In prHD, depressed mood, anxiety, and irritable behavior demonstrated good discriminative properties. In HD, depressed mood demonstrated a good relationship with the overall behavioral score. These data suggest that at least some UHDRS items appear to have utility across a broad range of severity, although many items demonstrate problematic features.
Url:
DOI: 10.1002/mds.23574
PubMed: 21370269
PubMed Central: 3157755
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Evans</name><affiliation wicri:level="1"><nlm:aff id="A1">OCBN, Toronto, Ontario, Canada</nlm:aff><country xml:lang="fr">Canada</country><wicri:regionArea>OCBN, Toronto, Ontario</wicri:regionArea><wicri:noRegion>Ontario</wicri:noRegion></affiliation></author></titleStmt><publicationStmt><idno type="wicri:source">PMC</idno><idno type="pmid">21370269</idno><idno type="pmc">3157755</idno><idno type="url">http://www.ncbi.nlm.nih.gov/pmc/articles/PMC3157755</idno><idno type="RBID">PMC:3157755</idno><idno type="doi">10.1002/mds.23574</idno><date when="2011">2011</date><idno type="wicri:Area/Pmc/Corpus">000173</idno><idno type="wicri:Area/Pmc/Curation">000173</idno><idno type="wicri:Area/Pmc/Checkpoint">000285</idno><idno type="wicri:source">PubMed</idno><idno type="wicri:Area/PubMed/Corpus">001414</idno><idno type="wicri:Area/PubMed/Curation">001414</idno><idno type="wicri:Area/PubMed/Checkpoint">001477</idno><idno type="wicri:Area/Ncbi/Merge">003009</idno><idno type="wicri:Area/Ncbi/Curation">003009</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en" level="a" type="main">An Item Response Analysis of the Motor and Behavioral Subscales of the Unified Huntington's Disease Rating Scale in Huntington Disease Gene Expansion Carriers</title><author><name sortKey="Vaccarino, Anthony L" sort="Vaccarino, Anthony L" uniqKey="Vaccarino A" first="Anthony L." last="Vaccarino">Anthony L. Vaccarino</name><affiliation wicri:level="1"><nlm:aff id="A1">OCBN, Toronto, Ontario, Canada</nlm:aff><country xml:lang="fr">Canada</country><wicri:regionArea>OCBN, Toronto, Ontario</wicri:regionArea><wicri:noRegion>Ontario</wicri:noRegion></affiliation></author><author><name sortKey="Anderson, Karen" sort="Anderson, Karen" uniqKey="Anderson K" first="Karen" last="Anderson">Karen Anderson</name><affiliation wicri:level="2"><nlm:aff id="A2">Department of Psychiatry, University of Maryland School of Medicine, Baltimore, Maryland, USA</nlm:aff><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Psychiatry, University of Maryland School of Medicine, Baltimore, Maryland</wicri:regionArea><placeName><region type="state">Maryland</region></placeName></affiliation></author><author><name sortKey="Borowsky, Beth" sort="Borowsky, Beth" uniqKey="Borowsky B" first="Beth" last="Borowsky">Beth Borowsky</name><affiliation wicri:level="2"><nlm:aff id="A3">CHDI Foundation, Princeton, New Jersey, USA</nlm:aff><country xml:lang="fr">États-Unis</country><wicri:regionArea>CHDI Foundation, Princeton, 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="2"><nlm:aff id="A4">Department of Neurology, University of Utah School of Medicine, Salt Lake City, Utah, USA</nlm:aff><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Neurology, University of Utah School of Medicine, Salt Lake City, Utah</wicri:regionArea><placeName><region type="state">Utah</region></placeName></affiliation></author><author><name sortKey="Giuliano, Joseph" sort="Giuliano, Joseph" uniqKey="Giuliano J" first="Joseph" last="Giuliano">Joseph Giuliano</name><affiliation wicri:level="2"><nlm:aff id="A3">CHDI Foundation, Princeton, New Jersey, USA</nlm:aff><country xml:lang="fr">États-Unis</country><wicri:regionArea>CHDI Foundation, Princeton, New Jersey</wicri:regionArea><placeName><region type="state">New Jersey</region></placeName></affiliation></author><author><name sortKey="Guttman, Mark" sort="Guttman, Mark" uniqKey="Guttman M" first="Mark" last="Guttman">Mark Guttman</name><affiliation wicri:level="1"><nlm:aff id="A5">The Center for Movement Disorders, Markham, Ontario, Canada</nlm:aff><country xml:lang="fr">Canada</country><wicri:regionArea>The Center for Movement Disorders, Markham, Ontario</wicri:regionArea><wicri:noRegion>Ontario</wicri:noRegion></affiliation></author><author><name sortKey="Ho, Aileen K" sort="Ho, Aileen K" uniqKey="Ho A" first="Aileen K." last="Ho">Aileen K. Ho</name><affiliation wicri:level="1"><nlm:aff id="A6">School of Psychology and Clinical Language Sciences, University of Reading, Reading, UK</nlm:aff><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>School of Psychology and Clinical Language Sciences, University of Reading, Reading</wicri:regionArea><wicri:noRegion>Reading</wicri:noRegion></affiliation></author><author><name sortKey="Orth, Michael" sort="Orth, Michael" uniqKey="Orth M" first="Michael" last="Orth">Michael Orth</name><affiliation wicri:level="4"><nlm:aff id="A7">EHDN, University of Ulm, Ulm, Germany</nlm:aff><country xml:lang="fr">Allemagne</country><wicri:regionArea>EHDN, University of Ulm, Ulm</wicri:regionArea><placeName><region type="land" nuts="1">Bade-Wurtemberg</region><region type="district" nuts="2">District de Tübingen</region><settlement type="city">Ulm</settlement></placeName><orgName type="university">Université d'Ulm</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"><nlm:aff id="A8">Department of Psychiatry, University of Iowa, Iowa City, USA</nlm:aff><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Psychiatry, University of Iowa, Iowa City</wicri:regionArea><orgName type="university">Université de l'Iowa</orgName><placeName><settlement type="city">Iowa City</settlement><region type="state">Iowa</region></placeName></affiliation></author><author><name sortKey="Sills, Terrence" sort="Sills, Terrence" uniqKey="Sills T" first="Terrence" last="Sills">Terrence Sills</name><affiliation wicri:level="1"><nlm:aff id="A1">OCBN, Toronto, Ontario, Canada</nlm:aff><country xml:lang="fr">Canada</country><wicri:regionArea>OCBN, Toronto, Ontario</wicri:regionArea><wicri:noRegion>Ontario</wicri:noRegion></affiliation></author><author><name sortKey="Van Kammen, Daniel P" sort="Van Kammen, Daniel P" uniqKey="Van Kammen D" first="Daniel P." last="Van Kammen">Daniel P. Van Kammen</name><affiliation wicri:level="2"><nlm:aff id="A3">CHDI Foundation, Princeton, New Jersey, USA</nlm:aff><country xml:lang="fr">États-Unis</country><wicri:regionArea>CHDI Foundation, Princeton, New Jersey</wicri:regionArea><placeName><region type="state">New Jersey</region></placeName></affiliation></author><author><name sortKey="Evans, Kenneth R" sort="Evans, Kenneth R" uniqKey="Evans K" first="Kenneth R." last="Evans">Kenneth R. Evans</name><affiliation wicri:level="1"><nlm:aff id="A1">OCBN, Toronto, Ontario, Canada</nlm:aff><country xml:lang="fr">Canada</country><wicri:regionArea>OCBN, Toronto, Ontario</wicri:regionArea><wicri:noRegion>Ontario</wicri:noRegion></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="2011">2011</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adolescent</term><term>Adult</term><term>Aged</term><term>Aged, 80 and over</term><term>Behavioral Symptoms (etiology)</term><term>Disability Evaluation</term><term>Female</term><term>Humans</term><term>Huntington Disease (complications)</term><term>Huntington Disease (diagnosis)</term><term>Huntington Disease (genetics)</term><term>Male</term><term>Middle Aged</term><term>Movement Disorders (etiology)</term><term>Severity of Illness Index</term><term>Trinucleotide Repeat Expansion (genetics)</term><term>Young Adult</term></keywords><keywords scheme="MESH" qualifier="complications" xml:lang="en"><term>Huntington Disease</term></keywords><keywords scheme="MESH" qualifier="diagnosis" xml:lang="en"><term>Huntington Disease</term></keywords><keywords scheme="MESH" qualifier="etiology" xml:lang="en"><term>Behavioral Symptoms</term><term>Movement Disorders</term></keywords><keywords scheme="MESH" qualifier="genetics" xml:lang="en"><term>Huntington Disease</term><term>Trinucleotide Repeat Expansion</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adolescent</term><term>Adult</term><term>Aged</term><term>Aged, 80 and over</term><term>Disability Evaluation</term><term>Female</term><term>Humans</term><term>Male</term><term>Middle Aged</term><term>Severity of Illness Index</term><term>Young Adult</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en"><p id="P1">Although the Unified Huntington's Disease Rating Scale (UHDRS) is widely used in the assessment of Huntington disease (HD), the ability of individual items to discriminate individual differences in motor or behavioral manifestations has not been extensively studied in HD gene expansion carriers without a motor-defined clinical diagnosis (i.e., prodromal-HD or prHD). To elucidate the relationship between scores on individual motor and behavioral UHDRS items and total score for each subscale, a non-parametric item response analysis was performed on retrospective data from two multicentre, longitudinal studies. Motor and Behavioral assessments were supplied for 737 prHD individuals with data from 2114 visits (PREDICT-HD) and 686 HD individuals with data from 1482 visits (REGISTRY). Option characteristic curves were generated for UHDRS subscale items in relation to their subscale score. In prHD, overall severity of motor signs was low and participants had scores of 2 or above on very few items. In HD, motor items that assessed ocular pursuit, saccade initiation, finger tapping, tandem walking, and to a lesser extent saccade velocity, dysarthia, tongue protrusion, pronation/supination, Luria, bradykinesia, choreas, gait and balance on the retropulsion test were found to discriminate individual differences across a broad range of motor severity. In prHD, depressed mood, anxiety, and irritable behavior demonstrated good discriminative properties. In HD, depressed mood demonstrated a good relationship with the overall behavioral score. These data suggest that at least some UHDRS items appear to have utility across a broad range of severity, although many items demonstrate problematic features.</p></div></front></TEI></record>Pour manipuler ce document sous Unix (Dilib)
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