Reliability and validity of the international cooperative ataxia rating scale : A study in 156 spinocerebellar ataxia patients
Identifieur interne : 003632 ( Main/Curation ); précédent : 003631; suivant : 003633Reliability and validity of the international cooperative ataxia rating scale : A study in 156 spinocerebellar ataxia patients
Auteurs : Tanja Schmitz-Hübsch [Allemagne] ; Sophie Tezenas Du Montcel [France] ; Laszlo Baliko [Hongrie] ; Sylvia Boesch [Autriche] ; Sara Bonato [Belgique] ; Roberto Fancellu [Italie] ; Paola Giunti [Royaume-Uni] ; Christoph Globas [Allemagne] ; Jun-Suk Kang [Allemagne] ; Berry Kremer [Pays-Bas] ; Caterina Mariotti [Italie] ; Bela Melegh [Hongrie] ; Maryla Rakowicz [Pologne] ; Rafal Rola [Pologne] ; Sylvie Romano [Italie] ; Lodger Schols [Allemagne] ; Sandra Szymanski [Allemagne] ; Bart P. C. Van De Warrenburg [Pays-Bas] ; Elzbieta Zdzienicka [Pologne] ; Alexandra Dürr [France] ; Thomas Klockgether [Allemagne]Source :
- Movement disorders [ 0885-3185 ] ; 2006.
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Abstract
To evaluate the efficacy of treatments in spinocerebellar ataxias (SCAs), appropriate clinical scales are required. This study evaluated metric properties of the International Cooperative Ataxia Rating Scale (ICARS) in 156 SCA patients and 8 controls. ICARS was found to be a reliable scale satisfying accepted criteria for interrater reliability, test-retest reliability, and internal consistency. Although validity testing was limited, we found evidence of validity of ICARS when ataxia disease stages and Barthel index were used as external criteria. On the other hand, our study revealed two major problems associated with the use of ICARS. First, the redundant and overlapping nature of several items gave rise to a considerable number of contradictory ratings. Second, a factorial analysis showed that the rating results were determined by four different factors that did not coincide with the ICARS subscales, thus questioning the justification of ICARS subscore analysis in clinical trials.
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<author><name sortKey="Durr, Alexandra" sort="Durr, Alexandra" uniqKey="Durr A" first="Alexandra" last="Dürr">Alexandra Dürr</name>
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<author><name sortKey="Klockgether, Thomas" sort="Klockgether, Thomas" uniqKey="Klockgether T" first="Thomas" last="Klockgether">Thomas Klockgether</name>
<affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Department of Neurology, University Hospital of Bonn</s1>
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<s3>DEU</s3>
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<author><name sortKey="Bonato, Sara" sort="Bonato, Sara" uniqKey="Bonato S" first="Sara" last="Bonato">Sara Bonato</name>
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<affiliation wicri:level="3"><inist:fA14 i1="07"><s1>Department of Biochemistry and Genetics, Istituto Nazionale Neurologico C. Besta</s1>
<s2>Milan</s2>
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<sZ>6 aut.</sZ>
<sZ>11 aut.</sZ>
<sZ>15 aut.</sZ>
</inist:fA14>
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<placeName><settlement type="city">Milan</settlement>
<region nuts="2">Lombardie</region>
</placeName>
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<author><name sortKey="Giunti, Paola" sort="Giunti, Paola" uniqKey="Giunti P" first="Paola" last="Giunti">Paola Giunti</name>
<affiliation wicri:level="3"><inist:fA14 i1="08"><s1>Institute of Neurology</s1>
<s2>London</s2>
<s3>GBR</s3>
<sZ>7 aut.</sZ>
</inist:fA14>
<country>Royaume-Uni</country>
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<region type="country">Angleterre</region>
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</placeName>
</affiliation>
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<author><name sortKey="Globas, Christoph" sort="Globas, Christoph" uniqKey="Globas C" first="Christoph" last="Globas">Christoph Globas</name>
<affiliation wicri:level="3"><inist:fA14 i1="09"><s1>Department of Neurology and Hertie Institute for Clinical Brain Research, University of Tübingen</s1>
<s2>Tübingen</s2>
<s3>DEU</s3>
<sZ>8 aut.</sZ>
<sZ>16 aut.</sZ>
</inist:fA14>
<country>Allemagne</country>
<placeName><region type="land" nuts="1">Bade-Wurtemberg</region>
<region type="district" nuts="2">District de Tübingen</region>
<settlement type="city">Tübingen</settlement>
</placeName>
</affiliation>
</author>
<author><name sortKey="Kang, Jun Suk" sort="Kang, Jun Suk" uniqKey="Kang J" first="Jun-Suk" last="Kang">Jun-Suk Kang</name>
<affiliation wicri:level="1"><inist:fA14 i1="10"><s1>Department of Neurology, University of Frankfurt</s1>
<s2>Frankfurt</s2>
<s3>DEU</s3>
<sZ>9 aut.</sZ>
</inist:fA14>
<country>Allemagne</country>
<wicri:noRegion>Frankfurt</wicri:noRegion>
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<wicri:noRegion>Frankfurt</wicri:noRegion>
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</author>
<author><name sortKey="Kremer, Berry" sort="Kremer, Berry" uniqKey="Kremer B" first="Berry" last="Kremer">Berry Kremer</name>
<affiliation wicri:level="3"><inist:fA14 i1="11"><s1>Department of Neurology, Radboud University Nijmegen Medical Center</s1>
<s2>Nijmegen</s2>
<s3>NLD</s3>
<sZ>10 aut.</sZ>
<sZ>18 aut.</sZ>
</inist:fA14>
<country>Pays-Bas</country>
<placeName><settlement type="city">Nimègue</settlement>
<region type="province" nuts="2">Gueldre</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Mariotti, Caterina" sort="Mariotti, Caterina" uniqKey="Mariotti C" first="Caterina" last="Mariotti">Caterina Mariotti</name>
<affiliation wicri:level="3"><inist:fA14 i1="07"><s1>Department of Biochemistry and Genetics, Istituto Nazionale Neurologico C. Besta</s1>
<s2>Milan</s2>
<s3>ITA</s3>
<sZ>6 aut.</sZ>
<sZ>11 aut.</sZ>
<sZ>15 aut.</sZ>
</inist:fA14>
<country>Italie</country>
<placeName><settlement type="city">Milan</settlement>
<region nuts="2">Lombardie</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Melegh, Bela" sort="Melegh, Bela" uniqKey="Melegh B" first="Bela" last="Melegh">Bela Melegh</name>
<affiliation wicri:level="1"><inist:fA14 i1="12"><s1>Department of Genetics, University of Pecs</s1>
<s2>Pecs</s2>
<s3>HUN</s3>
<sZ>12 aut.</sZ>
</inist:fA14>
<country>Hongrie</country>
<wicri:noRegion>Pecs</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Rakowicz, Maryla" sort="Rakowicz, Maryla" uniqKey="Rakowicz M" first="Maryla" last="Rakowicz">Maryla Rakowicz</name>
<affiliation wicri:level="1"><inist:fA14 i1="13"><s1>Institute of Psychiatry and Neurology</s1>
<s2>Warsaw</s2>
<s3>POL</s3>
<sZ>13 aut.</sZ>
<sZ>14 aut.</sZ>
<sZ>19 aut.</sZ>
</inist:fA14>
<country>Pologne</country>
<wicri:noRegion>Institute of Psychiatry and Neurology</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Rola, Rafal" sort="Rola, Rafal" uniqKey="Rola R" first="Rafal" last="Rola">Rafal Rola</name>
<affiliation wicri:level="1"><inist:fA14 i1="13"><s1>Institute of Psychiatry and Neurology</s1>
<s2>Warsaw</s2>
<s3>POL</s3>
<sZ>13 aut.</sZ>
<sZ>14 aut.</sZ>
<sZ>19 aut.</sZ>
</inist:fA14>
<country>Pologne</country>
<wicri:noRegion>Institute of Psychiatry and Neurology</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Romano, Sylvie" sort="Romano, Sylvie" uniqKey="Romano S" first="Sylvie" last="Romano">Sylvie Romano</name>
<affiliation wicri:level="3"><inist:fA14 i1="07"><s1>Department of Biochemistry and Genetics, Istituto Nazionale Neurologico C. Besta</s1>
<s2>Milan</s2>
<s3>ITA</s3>
<sZ>6 aut.</sZ>
<sZ>11 aut.</sZ>
<sZ>15 aut.</sZ>
</inist:fA14>
<country>Italie</country>
<placeName><settlement type="city">Milan</settlement>
<region nuts="2">Lombardie</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Schols, Lodger" sort="Schols, Lodger" uniqKey="Schols L" first="Lodger" last="Schols">Lodger Schols</name>
<affiliation wicri:level="3"><inist:fA14 i1="09"><s1>Department of Neurology and Hertie Institute for Clinical Brain Research, University of Tübingen</s1>
<s2>Tübingen</s2>
<s3>DEU</s3>
<sZ>8 aut.</sZ>
<sZ>16 aut.</sZ>
</inist:fA14>
<country>Allemagne</country>
<placeName><region type="land" nuts="1">Bade-Wurtemberg</region>
<region type="district" nuts="2">District de Tübingen</region>
<settlement type="city">Tübingen</settlement>
</placeName>
</affiliation>
</author>
<author><name sortKey="Szymanski, Sandra" sort="Szymanski, Sandra" uniqKey="Szymanski S" first="Sandra" last="Szymanski">Sandra Szymanski</name>
<affiliation wicri:level="1"><inist:fA14 i1="14"><s1>Department of Neurology, St. Josef Hospital, University Hospital</s1>
<s2>Bochum</s2>
<s3>DEU</s3>
<sZ>17 aut.</sZ>
</inist:fA14>
<country>Allemagne</country>
<wicri:noRegion>Bochum</wicri:noRegion>
<wicri:noRegion>University Hospital</wicri:noRegion>
<wicri:noRegion>Bochum</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Van De Warrenburg, Bart P C" sort="Van De Warrenburg, Bart P C" uniqKey="Van De Warrenburg B" first="Bart P. C." last="Van De Warrenburg">Bart P. C. Van De Warrenburg</name>
<affiliation wicri:level="3"><inist:fA14 i1="11"><s1>Department of Neurology, Radboud University Nijmegen Medical Center</s1>
<s2>Nijmegen</s2>
<s3>NLD</s3>
<sZ>10 aut.</sZ>
<sZ>18 aut.</sZ>
</inist:fA14>
<country>Pays-Bas</country>
<placeName><settlement type="city">Nimègue</settlement>
<region type="province" nuts="2">Gueldre</region>
</placeName>
</affiliation>
</author>
<author><name sortKey="Zdzienicka, Elzbieta" sort="Zdzienicka, Elzbieta" uniqKey="Zdzienicka E" first="Elzbieta" last="Zdzienicka">Elzbieta Zdzienicka</name>
<affiliation wicri:level="1"><inist:fA14 i1="13"><s1>Institute of Psychiatry and Neurology</s1>
<s2>Warsaw</s2>
<s3>POL</s3>
<sZ>13 aut.</sZ>
<sZ>14 aut.</sZ>
<sZ>19 aut.</sZ>
</inist:fA14>
<country>Pologne</country>
<wicri:noRegion>Institute of Psychiatry and Neurology</wicri:noRegion>
</affiliation>
</author>
<author><name sortKey="Durr, Alexandra" sort="Durr, Alexandra" uniqKey="Durr A" first="Alexandra" last="Dürr">Alexandra Dürr</name>
<affiliation wicri:level="1"><inist:fA14 i1="15"><s1>INSERM U679 and Department of Genetics, Cytogenetics and Embryology, Hôpital de la Pitié-Salpêtrière</s1>
<s2>Paris</s2>
<s3>FRA</s3>
<sZ>20 aut.</sZ>
</inist:fA14>
<country>France</country>
<placeName><settlement type="city">Paris</settlement>
</placeName>
</affiliation>
</author>
<author><name sortKey="Klockgether, Thomas" sort="Klockgether, Thomas" uniqKey="Klockgether T" first="Thomas" last="Klockgether">Thomas Klockgether</name>
<affiliation wicri:level="3"><inist:fA14 i1="01"><s1>Department of Neurology, University Hospital of Bonn</s1>
<s2>Bonn</s2>
<s3>DEU</s3>
<sZ>1 aut.</sZ>
<sZ>21 aut.</sZ>
</inist:fA14>
<country>Allemagne</country>
<placeName><region type="land" nuts="1">Rhénanie-du-Nord-Westphalie</region>
<region type="district" nuts="2">District de Cologne</region>
<settlement type="city">Bonn</settlement>
</placeName>
</affiliation>
</author>
</analytic>
<series><title level="j" type="main">Movement disorders</title>
<title level="j" type="abbreviated">Mov. disord.</title>
<idno type="ISSN">0885-3185</idno>
<imprint><date when="2006">2006</date>
</imprint>
</series>
</biblStruct>
</sourceDesc>
<seriesStmt><title level="j" type="main">Movement disorders</title>
<title level="j" type="abbreviated">Mov. disord.</title>
<idno type="ISSN">0885-3185</idno>
</seriesStmt>
</fileDesc>
<profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Evaluation scale</term>
<term>Human</term>
<term>Nervous system diseases</term>
<term>Reliability</term>
<term>Spinocerebellar ataxia</term>
<term>Validity</term>
</keywords>
<keywords scheme="Pascal" xml:lang="fr"><term>Système nerveux pathologie</term>
<term>Fiabilité</term>
<term>Validité</term>
<term>Ataxie spinocérébelleuse</term>
<term>Echelle d'évaluation</term>
<term>Homme</term>
</keywords>
<keywords scheme="Wicri" type="topic" xml:lang="fr"><term>Homme</term>
</keywords>
</textClass>
</profileDesc>
</teiHeader>
<front><div type="abstract" xml:lang="en">To evaluate the efficacy of treatments in spinocerebellar ataxias (SCAs), appropriate clinical scales are required. This study evaluated metric properties of the International Cooperative Ataxia Rating Scale (ICARS) in 156 SCA patients and 8 controls. ICARS was found to be a reliable scale satisfying accepted criteria for interrater reliability, test-retest reliability, and internal consistency. Although validity testing was limited, we found evidence of validity of ICARS when ataxia disease stages and Barthel index were used as external criteria. On the other hand, our study revealed two major problems associated with the use of ICARS. First, the redundant and overlapping nature of several items gave rise to a considerable number of contradictory ratings. Second, a factorial analysis showed that the rating results were determined by four different factors that did not coincide with the ICARS subscales, thus questioning the justification of ICARS subscore analysis in clinical trials.</div>
</front>
</TEI>
</record>
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