Progression of multiple system atrophy (MSA): A prospective natural history study by the European MSA Study Group (EMSA SG)
Identifieur interne : 001221 ( Main/Exploration ); précédent : 001220; suivant : 001222Progression of multiple system atrophy (MSA): A prospective natural history study by the European MSA Study Group (EMSA SG)
Auteurs : Felix Geser [Autriche] ; Gregor K. Wenning [Autriche] ; Klaus Seppi [Autriche] ; Michaela Stampfer-Kountchev [Autriche] ; Christoph Scherfler [Autriche] ; Martin Sawires [Autriche] ; Carolin Frick [Autriche] ; Jean-Pierre Ndayisaba [Autriche] ; Hanno Ulmer [Autriche] ; Maria T. Pellecchia [Italie] ; Paolo Barone [Italie] ; Hee T. Kim [Royaume-Uni] ; Juzar Hooker [Royaume-Uni] ; Niall P. Quinn [Royaume-Uni] ; Adriana Cardozo [Espagne] ; Eduardo Tolosa [Espagne] ; Michael Abele [Allemagne] ; Thomas Klockgether [Allemagne] ; Karen Stergaard [Danemark] ; Erik Dupont [Danemark] ; Nicole Schimke [Allemagne] ; Karla M. Eggert [Allemagne] ; Wolfgang Oertel [Allemagne] ; Ruth Djaldetti [Israël] ; Werner Poewe [Autriche]Source :
- Movement Disorders [ 0885-3185 ] ; 2006-02.
English descriptors
Abstract
The disease‐specific Unified Multiple System Atrophy Rating Scale (UMSARS) has been developed recently and validated for assessing disease severity in multiple system atrophy (MSA). Here, we aimed at (1) assessing rates of disease progression in MSA and (2) validating UMSARS for sensitivity to change over time. Impairment was assessed at two time points 12 months apart using UMSARS Part I (historical review), UMSARS Part II (motor examination), as well as measures of global disease severity, including UMSARS Part IV, Hoehn and Yahr (HY) Parkinson's disease staging, Schwab England Activities of Daily Living (SE ADL), and a three‐point global Severity Scale (SS3). Fifty patients (male:female ratio, 1:0.9; possible MSA, 16%; probable MSA, 84%; MSA‐parkinsonian, 58%; MSA‐cerebellar, 42%) were assessed twice with an interval of 12.3 months. UMSARS II scores progressed by 57.3% (P < 0.0001) and UMSARS I scores by 35.6% (P < 0.0001) in relation to the respective baseline scores with no differences between motor subtypes, diagnostic categories and gender. Significant inverse correlations between (1) UMSARS I or UMSARS II progression and (2) baseline disability measures (i.e., the respective UMSARS or SS3 scores) and disease duration were found. Furthermore, the increases in HY staging, SE ADL and SS3 correlated significantly with UMSARS I, UMSARS II, and UMSARS IV progression. This report is the first prospective study showing rapid annual UMSARS rates of decline in MSA. Our data contribute to the ongoing validation process of UMSARS, and they facilitate the planning and implementation of future neuroprotective intervention trials. © 2005 Movement Disorder Society
Url:
DOI: 10.1002/mds.20678
Affiliations:
- Allemagne, Autriche, Danemark, Espagne, Israël, Italie, Royaume-Uni
- Angleterre, Grand Londres, Tyrol (Land)
- Innsbruck, Londres
- Université de médecine d'Innsbruck
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Wenning</name></author><author><name sortKey="Seppi, Klaus" sort="Seppi, Klaus" uniqKey="Seppi K" first="Klaus" last="Seppi">Klaus Seppi</name></author><author><name sortKey="Stampfer Ountchev, Michaela" sort="Stampfer Ountchev, Michaela" uniqKey="Stampfer Ountchev M" first="Michaela" last="Stampfer-Kountchev">Michaela Stampfer-Kountchev</name></author><author><name sortKey="Scherfler, Christoph" sort="Scherfler, Christoph" uniqKey="Scherfler C" first="Christoph" last="Scherfler">Christoph Scherfler</name></author><author><name sortKey="Sawires, Martin" sort="Sawires, Martin" uniqKey="Sawires M" first="Martin" last="Sawires">Martin Sawires</name></author><author><name sortKey="Frick, Carolin" sort="Frick, Carolin" uniqKey="Frick C" first="Carolin" last="Frick">Carolin Frick</name></author><author><name sortKey="Ndayisaba, Jean Ierre" sort="Ndayisaba, Jean Ierre" uniqKey="Ndayisaba J" first="Jean-Pierre" last="Ndayisaba">Jean-Pierre Ndayisaba</name></author><author><name sortKey="Ulmer, Hanno" sort="Ulmer, Hanno" uniqKey="Ulmer H" first="Hanno" last="Ulmer">Hanno Ulmer</name></author><author><name sortKey="Pellecchia, Maria T" sort="Pellecchia, Maria T" uniqKey="Pellecchia M" first="Maria T." last="Pellecchia">Maria T. 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Pellecchia</name><affiliation wicri:level="1"><country xml:lang="fr">Italie</country><wicri:regionArea>Department of Neurological Sciences, University Federico II, Naples</wicri:regionArea><wicri:noRegion>Naples</wicri:noRegion></affiliation></author><author><name sortKey="Barone, Paolo" sort="Barone, Paolo" uniqKey="Barone P" first="Paolo" last="Barone">Paolo Barone</name><affiliation wicri:level="1"><country xml:lang="fr">Italie</country><wicri:regionArea>Department of Neurological Sciences, University Federico II, Naples</wicri:regionArea><wicri:noRegion>Naples</wicri:noRegion></affiliation></author><author><name sortKey="Kim, Hee T" sort="Kim, Hee T" uniqKey="Kim H" first="Hee T." last="Kim">Hee T. Kim</name><affiliation wicri:level="3"><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>University College London, Institute of Neurology, Queen Square, London</wicri:regionArea><placeName><settlement type="city">Londres</settlement><region type="country">Angleterre</region><region type="région" nuts="1">Grand Londres</region></placeName></affiliation></author><author><name sortKey="Hooker, Juzar" sort="Hooker, Juzar" uniqKey="Hooker J" first="Juzar" last="Hooker">Juzar Hooker</name><affiliation wicri:level="3"><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>University College London, Institute of Neurology, Queen Square, London</wicri:regionArea><placeName><settlement type="city">Londres</settlement><region type="country">Angleterre</region><region type="région" nuts="1">Grand Londres</region></placeName></affiliation></author><author><name sortKey="Quinn, Niall P" sort="Quinn, Niall P" uniqKey="Quinn N" first="Niall P." last="Quinn">Niall P. Quinn</name><affiliation wicri:level="3"><country xml:lang="fr">Royaume-Uni</country><wicri:regionArea>University College London, Institute of Neurology, Queen Square, London</wicri:regionArea><placeName><settlement type="city">Londres</settlement><region type="country">Angleterre</region><region type="région" nuts="1">Grand Londres</region></placeName></affiliation></author><author><name sortKey="Cardozo, Adriana" sort="Cardozo, Adriana" uniqKey="Cardozo A" first="Adriana" last="Cardozo">Adriana Cardozo</name><affiliation wicri:level="1"><country xml:lang="fr">Espagne</country><wicri:regionArea>Universitat de Barcelona, Hospital Clinic, Department of Neurology</wicri:regionArea><wicri:noRegion>Department of Neurology</wicri:noRegion></affiliation></author><author><name sortKey="Tolosa, Eduardo" sort="Tolosa, Eduardo" uniqKey="Tolosa E" first="Eduardo" last="Tolosa">Eduardo Tolosa</name><affiliation wicri:level="1"><country xml:lang="fr">Espagne</country><wicri:regionArea>Universitat de Barcelona, Hospital Clinic, Department of Neurology</wicri:regionArea><wicri:noRegion>Department of Neurology</wicri:noRegion></affiliation></author><author><name sortKey="Abele, Michael" sort="Abele, Michael" uniqKey="Abele M" first="Michael" last="Abele">Michael Abele</name><affiliation wicri:level="1"><country xml:lang="fr">Allemagne</country><wicri:regionArea>University of Bonn, Department of Neurology</wicri:regionArea><wicri:noRegion>Department of Neurology</wicri:noRegion><wicri:noRegion>Department of Neurology</wicri:noRegion></affiliation></author><author><name sortKey="Klockgether, Thomas" sort="Klockgether, Thomas" uniqKey="Klockgether T" first="Thomas" last="Klockgether">Thomas Klockgether</name><affiliation wicri:level="1"><country xml:lang="fr">Allemagne</country><wicri:regionArea>University of Bonn, Department of Neurology</wicri:regionArea><wicri:noRegion>Department of Neurology</wicri:noRegion><wicri:noRegion>Department of Neurology</wicri:noRegion></affiliation></author><author><name sortKey=" Stergaard, Karen" sort=" Stergaard, Karen" uniqKey=" Stergaard K" first="Karen" last=" Stergaard">Karen Stergaard</name><affiliation wicri:level="1"><country xml:lang="fr">Danemark</country><wicri:regionArea>Aarhus Kummunehospital, Department of Neurology, Aarhus</wicri:regionArea><wicri:noRegion>Aarhus</wicri:noRegion></affiliation></author><author><name sortKey="Dupont, Erik" sort="Dupont, Erik" uniqKey="Dupont E" first="Erik" last="Dupont">Erik Dupont</name><affiliation wicri:level="1"><country xml:lang="fr">Danemark</country><wicri:regionArea>Aarhus Kummunehospital, Department of Neurology, Aarhus</wicri:regionArea><wicri:noRegion>Aarhus</wicri:noRegion></affiliation></author><author><name sortKey="Schimke, Nicole" sort="Schimke, Nicole" uniqKey="Schimke N" first="Nicole" last="Schimke">Nicole Schimke</name><affiliation wicri:level="1"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, Philipps‐University Marburg, Department of Neurology</wicri:regionArea><wicri:noRegion>Department of Neurology</wicri:noRegion><wicri:noRegion>Department of Neurology</wicri:noRegion></affiliation></author><author><name sortKey="Eggert, Karla M" sort="Eggert, Karla M" uniqKey="Eggert K" first="Karla M." last="Eggert">Karla M. Eggert</name><affiliation wicri:level="1"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, Philipps‐University Marburg, Department of Neurology</wicri:regionArea><wicri:noRegion>Department of Neurology</wicri:noRegion><wicri:noRegion>Department of Neurology</wicri:noRegion></affiliation></author><author><name sortKey="Oertel, Wolfgang" sort="Oertel, Wolfgang" uniqKey="Oertel W" first="Wolfgang" last="Oertel">Wolfgang Oertel</name><affiliation wicri:level="1"><country xml:lang="fr">Allemagne</country><wicri:regionArea>Department of Neurology, Philipps‐University Marburg, Department of Neurology</wicri:regionArea><wicri:noRegion>Department of Neurology</wicri:noRegion><wicri:noRegion>Department of Neurology</wicri:noRegion></affiliation></author><author><name sortKey="Djaldetti, Ruth" sort="Djaldetti, Ruth" uniqKey="Djaldetti R" first="Ruth" last="Djaldetti">Ruth Djaldetti</name><affiliation wicri:level="1"><country xml:lang="fr">Israël</country><wicri:regionArea>Department of Neurology, Rabin Medical Center, Petach‐Tiqva</wicri:regionArea><wicri:noRegion>Petach‐Tiqva</wicri:noRegion></affiliation></author><author><name sortKey="Poewe, Werner" sort="Poewe, Werner" uniqKey="Poewe W" first="Werner" last="Poewe">Werner Poewe</name><affiliation wicri:level="1"><country xml:lang="fr">Autriche</country><wicri:regionArea>Clinical Department of Neurology, Innsbruck Medical University</wicri:regionArea><wicri:noRegion>Innsbruck Medical University</wicri:noRegion><placeName><settlement type="city">Innsbruck</settlement><region nuts="2" type="region">Tyrol (Land)</region></placeName><orgName type="university">Université de médecine d'Innsbruck</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="2006-02">2006-02</date><biblScope unit="volume">21</biblScope><biblScope unit="issue">2</biblScope><biblScope unit="page" from="179">179</biblScope><biblScope unit="page" to="186">186</biblScope></imprint><idno type="ISSN">0885-3185</idno></series><idno type="istex">4F6EEEBBEF9136A65A1579FC89C1E049885C1002</idno><idno type="DOI">10.1002/mds.20678</idno><idno type="ArticleID">MDS20678</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Unified MSA Rating Scale</term><term>multiple system atrophy</term><term>progression</term><term>sensitivity to change</term></keywords></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">The disease‐specific Unified Multiple System Atrophy Rating Scale (UMSARS) has been developed recently and validated for assessing disease severity in multiple system atrophy (MSA). Here, we aimed at (1) assessing rates of disease progression in MSA and (2) validating UMSARS for sensitivity to change over time. Impairment was assessed at two time points 12 months apart using UMSARS Part I (historical review), UMSARS Part II (motor examination), as well as measures of global disease severity, including UMSARS Part IV, Hoehn and Yahr (HY) Parkinson's disease staging, Schwab England Activities of Daily Living (SE ADL), and a three‐point global Severity Scale (SS3). Fifty patients (male:female ratio, 1:0.9; possible MSA, 16%; probable MSA, 84%; MSA‐parkinsonian, 58%; MSA‐cerebellar, 42%) were assessed twice with an interval of 12.3 months. UMSARS II scores progressed by 57.3% (P < 0.0001) and UMSARS I scores by 35.6% (P < 0.0001) in relation to the respective baseline scores with no differences between motor subtypes, diagnostic categories and gender. Significant inverse correlations between (1) UMSARS I or UMSARS II progression and (2) baseline disability measures (i.e., the respective UMSARS or SS3 scores) and disease duration were found. Furthermore, the increases in HY staging, SE ADL and SS3 correlated significantly with UMSARS I, UMSARS II, and UMSARS IV progression. This report is the first prospective study showing rapid annual UMSARS rates of decline in MSA. Our data contribute to the ongoing validation process of UMSARS, and they facilitate the planning and implementation of future neuroprotective intervention trials. © 2005 Movement Disorder Society</div></front></TEI><affiliations><list><country><li>Allemagne</li><li>Autriche</li><li>Danemark</li><li>Espagne</li><li>Israël</li><li>Italie</li><li>Royaume-Uni</li></country><region><li>Angleterre</li><li>Grand Londres</li><li>Tyrol (Land)</li></region><settlement><li>Innsbruck</li><li>Londres</li></settlement><orgName><li>Université de médecine d'Innsbruck</li></orgName></list><tree><country name="Autriche"><noRegion><name sortKey="Geser, Felix" sort="Geser, Felix" uniqKey="Geser F" first="Felix" last="Geser">Felix Geser</name></noRegion><name sortKey="Frick, Carolin" sort="Frick, Carolin" uniqKey="Frick C" first="Carolin" last="Frick">Carolin Frick</name><name sortKey="Ndayisaba, Jean Ierre" sort="Ndayisaba, Jean Ierre" uniqKey="Ndayisaba J" first="Jean-Pierre" last="Ndayisaba">Jean-Pierre Ndayisaba</name><name sortKey="Poewe, Werner" sort="Poewe, Werner" uniqKey="Poewe W" first="Werner" last="Poewe">Werner Poewe</name><name sortKey="Sawires, Martin" sort="Sawires, Martin" uniqKey="Sawires M" first="Martin" last="Sawires">Martin Sawires</name><name sortKey="Scherfler, Christoph" sort="Scherfler, Christoph" uniqKey="Scherfler C" first="Christoph" last="Scherfler">Christoph Scherfler</name><name sortKey="Seppi, Klaus" sort="Seppi, Klaus" uniqKey="Seppi K" first="Klaus" last="Seppi">Klaus Seppi</name><name sortKey="Stampfer Ountchev, Michaela" sort="Stampfer Ountchev, Michaela" uniqKey="Stampfer Ountchev M" first="Michaela" last="Stampfer-Kountchev">Michaela Stampfer-Kountchev</name><name sortKey="Ulmer, Hanno" sort="Ulmer, Hanno" uniqKey="Ulmer H" first="Hanno" last="Ulmer">Hanno Ulmer</name><name sortKey="Wenning, Gregor K" sort="Wenning, Gregor K" uniqKey="Wenning G" first="Gregor K." last="Wenning">Gregor K. Wenning</name></country><country name="Italie"><noRegion><name sortKey="Pellecchia, Maria T" sort="Pellecchia, Maria T" uniqKey="Pellecchia M" first="Maria T." last="Pellecchia">Maria T. Pellecchia</name></noRegion><name sortKey="Barone, Paolo" sort="Barone, Paolo" uniqKey="Barone P" first="Paolo" last="Barone">Paolo Barone</name></country><country name="Royaume-Uni"><region name="Angleterre"><name sortKey="Kim, Hee T" sort="Kim, Hee T" uniqKey="Kim H" first="Hee T." last="Kim">Hee T. Kim</name></region><name sortKey="Hooker, Juzar" sort="Hooker, Juzar" uniqKey="Hooker J" first="Juzar" last="Hooker">Juzar Hooker</name><name sortKey="Quinn, Niall P" sort="Quinn, Niall P" uniqKey="Quinn N" first="Niall P." last="Quinn">Niall P. Quinn</name></country><country name="Espagne"><noRegion><name sortKey="Cardozo, Adriana" sort="Cardozo, Adriana" uniqKey="Cardozo A" first="Adriana" last="Cardozo">Adriana Cardozo</name></noRegion><name sortKey="Tolosa, Eduardo" sort="Tolosa, Eduardo" uniqKey="Tolosa E" first="Eduardo" last="Tolosa">Eduardo Tolosa</name></country><country name="Allemagne"><noRegion><name sortKey="Abele, Michael" sort="Abele, Michael" uniqKey="Abele M" first="Michael" last="Abele">Michael Abele</name></noRegion><name sortKey="Eggert, Karla M" sort="Eggert, Karla M" uniqKey="Eggert K" first="Karla M." last="Eggert">Karla M. Eggert</name><name sortKey="Klockgether, Thomas" sort="Klockgether, Thomas" uniqKey="Klockgether T" first="Thomas" last="Klockgether">Thomas Klockgether</name><name sortKey="Oertel, Wolfgang" sort="Oertel, Wolfgang" uniqKey="Oertel W" first="Wolfgang" last="Oertel">Wolfgang Oertel</name><name sortKey="Schimke, Nicole" sort="Schimke, Nicole" uniqKey="Schimke N" first="Nicole" last="Schimke">Nicole Schimke</name></country><country name="Danemark"><noRegion><name sortKey=" Stergaard, Karen" sort=" Stergaard, Karen" uniqKey=" Stergaard K" first="Karen" last=" Stergaard">Karen Stergaard</name></noRegion><name sortKey="Dupont, Erik" sort="Dupont, Erik" uniqKey="Dupont E" first="Erik" last="Dupont">Erik Dupont</name></country><country name="Israël"><noRegion><name sortKey="Djaldetti, Ruth" sort="Djaldetti, Ruth" uniqKey="Djaldetti R" first="Ruth" last="Djaldetti">Ruth Djaldetti</name></noRegion></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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