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 : 001414 ( PubMed/Curation ); précédent : 001413; suivant : 001415An 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 ; Beth Borowsky ; Kevin Duff ; Joseph Giuliano ; Mark Guttman ; Aileen K. Ho ; Michael Orth ; Jane S. Paulsen ; Terrence Sills ; Daniel P. Van Kammen ; Kenneth R. EvansSource :
- Movement disorders : official journal of the Movement Disorder Society [ 1531-8257 ] ; 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 (ie, prodromal-HD or prHD). To elucidate the relationship between scores on individual motor and behavioral UHDRS items and total score for each subscale, a nonparametric item response analysis was performed on retrospective data from 2 multicenter 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, dysarthria, 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.
DOI: 10.1002/mds.23574
PubMed: 21370269
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Ho</name></author><author><name sortKey="Orth, Michael" sort="Orth, Michael" uniqKey="Orth M" first="Michael" last="Orth">Michael Orth</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><author><name sortKey="Sills, Terrence" sort="Sills, Terrence" uniqKey="Sills T" first="Terrence" last="Sills">Terrence Sills</name></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></author><author><name sortKey="Evans, Kenneth R" sort="Evans, Kenneth R" uniqKey="Evans K" first="Kenneth R" last="Evans">Kenneth R. Evans</name></author></analytic><series><title level="j">Movement disorders : official journal of the Movement Disorder Society</title><idno type="eISSN">1531-8257</idno><imprint><date when="2011" type="published">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">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 (ie, prodromal-HD or prHD). To elucidate the relationship between scores on individual motor and behavioral UHDRS items and total score for each subscale, a nonparametric item response analysis was performed on retrospective data from 2 multicenter 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, dysarthria, 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.</div></front></TEI><pubmed><MedlineCitation Owner="NLM" Status="MEDLINE"><PMID Version="1">21370269</PMID><DateCreated><Year>2011</Year><Month>04</Month><Day>26</Day></DateCreated><DateCompleted><Year>2011</Year><Month>08</Month><Day>24</Day></DateCompleted><DateRevised><Year>2014</Year><Month>09</Month><Day>20</Day></DateRevised><Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1531-8257</ISSN><JournalIssue CitedMedium="Internet"><Volume>26</Volume><Issue>5</Issue><PubDate><Year>2011</Year><Month>Apr</Month></PubDate></JournalIssue><Title>Movement disorders : official journal of the Movement Disorder Society</Title><ISOAbbreviation>Mov. Disord.</ISOAbbreviation></Journal><ArticleTitle>An item response analysis of the motor and behavioral subscales of the unified Huntington's disease rating scale in huntington disease gene expansion carriers.</ArticleTitle><Pagination><MedlinePgn>877-84</MedlinePgn></Pagination><ELocationID EIdType="doi" ValidYN="Y">10.1002/mds.23574</ELocationID><Abstract><AbstractText>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 (ie, prodromal-HD or prHD). To elucidate the relationship between scores on individual motor and behavioral UHDRS items and total score for each subscale, a nonparametric item response analysis was performed on retrospective data from 2 multicenter 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, dysarthria, 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.</AbstractText><CopyrightInformation>Copyright © 2011 Movement Disorder Society.</CopyrightInformation></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Vaccarino</LastName><ForeName>Anthony L</ForeName><Initials>AL</Initials><AffiliationInfo><Affiliation>OCBN, Toronto, Ontario, Canada. avaccarino@ocbn.ca</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Anderson</LastName><ForeName>Karen</ForeName><Initials>K</Initials></Author><Author ValidYN="Y"><LastName>Borowsky</LastName><ForeName>Beth</ForeName><Initials>B</Initials></Author><Author ValidYN="Y"><LastName>Duff</LastName><ForeName>Kevin</ForeName><Initials>K</Initials></Author><Author ValidYN="Y"><LastName>Giuliano</LastName><ForeName>Joseph</ForeName><Initials>J</Initials></Author><Author ValidYN="Y"><LastName>Guttman</LastName><ForeName>Mark</ForeName><Initials>M</Initials></Author><Author ValidYN="Y"><LastName>Ho</LastName><ForeName>Aileen K</ForeName><Initials>AK</Initials></Author><Author ValidYN="Y"><LastName>Orth</LastName><ForeName>Michael</ForeName><Initials>M</Initials></Author><Author ValidYN="Y"><LastName>Paulsen</LastName><ForeName>Jane S</ForeName><Initials>JS</Initials></Author><Author ValidYN="Y"><LastName>Sills</LastName><ForeName>Terrence</ForeName><Initials>T</Initials></Author><Author ValidYN="Y"><LastName>van Kammen</LastName><ForeName>Daniel P</ForeName><Initials>DP</Initials></Author><Author ValidYN="Y"><LastName>Evans</LastName><ForeName>Kenneth R</ForeName><Initials>KR</Initials></Author><Author ValidYN="Y"><CollectiveName>PREDICT-HD and REGISTRY Investigators Coordinators</CollectiveName></Author></AuthorList><Language>eng</Language><GrantList CompleteYN="Y"><Grant><GrantID>R01 NS040068</GrantID><Acronym>NS</Acronym><Agency>NINDS NIH HHS</Agency><Country>United States</Country></Grant><Grant><GrantID>R01 NS040068-11</GrantID><Acronym>NS</Acronym><Agency>NINDS NIH HHS</Agency><Country>United States</Country></Grant></GrantList><PublicationTypeList><PublicationType UI="D016428">Journal Article</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2011</Year><Month>03</Month><Day>02</Day></ArticleDate></Article><MedlineJournalInfo><Country>United States</Country><MedlineTA>Mov Disord</MedlineTA><NlmUniqueID>8610688</NlmUniqueID><ISSNLinking>0885-3185</ISSNLinking></MedlineJournalInfo><CitationSubset>IM</CitationSubset><CommentsCorrectionsList><CommentsCorrections RefType="Cites"><RefSource>Arch Neurol. 2000 Sep;57(9):1326-30</RefSource><PMID Version="1">10987900</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Psychol Assess. 2001 Mar;13(1):127-39</RefSource><PMID Version="1">11281034</PMID></CommentsCorrections><CommentsCorrections 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Mar;11(2):136-42</RefSource><PMID Version="1">8684382</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Mov Disord. 1997 Mar;12(2):229-34</RefSource><PMID Version="1">9087982</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Am J Psychiatry. 2005 Apr;162(4):725-31</RefSource><PMID Version="1">15800145</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>J Sex Med. 2005 Nov;2(6):801-18</RefSource><PMID Version="1">16422805</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Arch Neurol. 2006 Jun;63(6):883-90</RefSource><PMID Version="1">16769871</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Eur Neuropsychopharmacol. 2006 Dec;16(8):601-11</RefSource><PMID Version="1">16769204</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Expert Opin Pharmacother. 2007 Feb;8(2):141-53</RefSource><PMID 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Version="1">18800370</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>J Neurol. 2008 Nov;255(11):1785-91</RefSource><PMID Version="1">19156490</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Lancet Neurol. 2009 Sep;8(9):791-801</RefSource><PMID Version="1">19646924</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Gait Posture. 2009 Oct;30(3):391-3</RefSource><PMID Version="1">19647432</PMID></CommentsCorrections><CommentsCorrections RefType="Cites"><RefSource>Mov Disord. 2009 Sep 15;24(12):1763-72</RefSource><PMID Version="1">19562761</PMID></CommentsCorrections></CommentsCorrectionsList><MeshHeadingList><MeshHeading><DescriptorName MajorTopicYN="N" UI="D000293">Adolescent</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D000328">Adult</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D000368">Aged</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D000369">Aged, 80 and over</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D001526">Behavioral Symptoms</DescriptorName><QualifierName MajorTopicYN="Y" UI="Q000209">etiology</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="Y" UI="D004185">Disability Evaluation</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D005260">Female</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D006801">Humans</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="Y" UI="D006816">Huntington Disease</DescriptorName><QualifierName MajorTopicYN="N" UI="Q000150">complications</QualifierName><QualifierName MajorTopicYN="N" UI="Q000175">diagnosis</QualifierName><QualifierName MajorTopicYN="N" UI="Q000235">genetics</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D008297">Male</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D008875">Middle Aged</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D009069">Movement Disorders</DescriptorName><QualifierName MajorTopicYN="Y" UI="Q000209">etiology</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="Y" UI="D012720">Severity of Illness Index</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D019680">Trinucleotide Repeat Expansion</DescriptorName><QualifierName MajorTopicYN="Y" UI="Q000235">genetics</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D055815">Young Adult</DescriptorName></MeshHeading></MeshHeadingList><OtherID Source="NLM">NIHMS316552</OtherID><OtherID Source="NLM">PMC3157755</OtherID></MedlineCitation><PubmedData><History><PubMedPubDate 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