Quantitative assessment of the evolution of cerebellar signs in spinocerebellar ataxias.
Identifieur interne : 001134 ( PubMed/Checkpoint ); précédent : 001133; suivant : 001135Quantitative assessment of the evolution of cerebellar signs in spinocerebellar ataxias.
Auteurs : Ellis Chan [France] ; Perrine Charles ; Pascale Ribai ; Cyril Goizet ; Cecilia Marelli ; Carlo-Maria Vincitorio ; Alice Le Bayon ; Lucie Guyant-Maréchal ; Nadia Vandenberghe ; Mathieu Anheim ; David Devos ; Leorah Freeman ; Isabelle Le Ber ; Karine N'Guyen ; Maya Tchikviladzé ; Pierre Labauge ; Didier Hannequin ; Alexis Brice ; Alexandra Durr ; Sophie Tezenas Du MontcelSource :
- Movement disorders : official journal of the Movement Disorder Society [ 1531-8257 ] ; 2011.
English descriptors
- KwdEn :
- Adult, Analysis of Variance, Cerebellum (pathology), Cerebellum (physiopathology), Disability Evaluation, Disease Progression, Female, Follow-Up Studies, Humans, Male, Middle Aged, Sample Size, Severity of Illness Index, Spinocerebellar Ataxias (diagnosis), Spinocerebellar Ataxias (physiopathology), Statistics as Topic, Young Adult.
- MESH :
- diagnosis : Spinocerebellar Ataxias.
- pathology : Cerebellum.
- physiopathology : Cerebellum, Spinocerebellar Ataxias.
- Adult, Analysis of Variance, Disability Evaluation, Disease Progression, Female, Follow-Up Studies, Humans, Male, Middle Aged, Sample Size, Severity of Illness Index, Statistics as Topic, Young Adult.
Abstract
Responsive ataxia rating scales are essential for determining outcome measures in clinical trials.
DOI: 10.1002/mds.23531
PubMed: 21287600
Affiliations:
Links toward previous steps (curation, corpus...)
Links to Exploration step
pubmed:21287600Le document en format XML
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last="Labauge">Pierre Labauge</name></author><author><name sortKey="Hannequin, Didier" sort="Hannequin, Didier" uniqKey="Hannequin D" first="Didier" last="Hannequin">Didier Hannequin</name></author><author><name sortKey="Brice, Alexis" sort="Brice, Alexis" uniqKey="Brice A" first="Alexis" last="Brice">Alexis Brice</name></author><author><name sortKey="Durr, Alexandra" sort="Durr, Alexandra" uniqKey="Durr A" first="Alexandra" last="Durr">Alexandra Durr</name></author><author><name sortKey="Du Montcel, Sophie Tezenas" sort="Du Montcel, Sophie Tezenas" uniqKey="Du Montcel S" first="Sophie Tezenas" last="Du Montcel">Sophie Tezenas Du Montcel</name></author></titleStmt><publicationStmt><idno type="wicri:source">PubMed</idno><date when="2011">2011</date><idno type="doi">10.1002/mds.23531</idno><idno type="RBID">pubmed:21287600</idno><idno type="pmid">21287600</idno><idno type="wicri:Area/PubMed/Corpus">001464</idno><idno type="wicri:Area/PubMed/Curation">001464</idno><idno type="wicri:Area/PubMed/Checkpoint">001134</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en">Quantitative assessment of the evolution of cerebellar signs in spinocerebellar ataxias.</title><author><name sortKey="Chan, Ellis" sort="Chan, Ellis" uniqKey="Chan E" first="Ellis" last="Chan">Ellis Chan</name><affiliation wicri:level="1"><nlm:affiliation>Department of Biostatistics and Medical Informatics and Pitié-Salpêtrière Charles-Foix Clinical Research Unit, AP-HP, Paris, France.</nlm:affiliation><country xml:lang="fr">France</country><wicri:regionArea>Department of Biostatistics and Medical Informatics and Pitié-Salpêtrière Charles-Foix Clinical Research Unit, AP-HP, Paris</wicri:regionArea><placeName><settlement type="city">Paris</settlement></placeName></affiliation></author><author><name sortKey="Charles, Perrine" sort="Charles, Perrine" uniqKey="Charles P" first="Perrine" last="Charles">Perrine Charles</name></author><author><name sortKey="Ribai, Pascale" sort="Ribai, Pascale" uniqKey="Ribai P" first="Pascale" last="Ribai">Pascale Ribai</name></author><author><name sortKey="Goizet, Cyril" sort="Goizet, Cyril" uniqKey="Goizet C" first="Cyril" last="Goizet">Cyril Goizet</name></author><author><name sortKey="Marelli, Cecilia" sort="Marelli, Cecilia" uniqKey="Marelli C" first="Cecilia" last="Marelli">Cecilia Marelli</name></author><author><name sortKey="Vincitorio, Carlo Maria" sort="Vincitorio, Carlo Maria" uniqKey="Vincitorio C" first="Carlo-Maria" last="Vincitorio">Carlo-Maria Vincitorio</name></author><author><name sortKey="Le Bayon, Alice" sort="Le Bayon, Alice" uniqKey="Le Bayon A" first="Alice" last="Le Bayon">Alice Le Bayon</name></author><author><name sortKey="Guyant Marechal, Lucie" sort="Guyant Marechal, Lucie" uniqKey="Guyant Marechal L" first="Lucie" last="Guyant-Maréchal">Lucie Guyant-Maréchal</name></author><author><name sortKey="Vandenberghe, Nadia" sort="Vandenberghe, Nadia" uniqKey="Vandenberghe N" first="Nadia" last="Vandenberghe">Nadia Vandenberghe</name></author><author><name sortKey="Anheim, Mathieu" sort="Anheim, Mathieu" uniqKey="Anheim M" first="Mathieu" last="Anheim">Mathieu Anheim</name></author><author><name sortKey="Devos, David" sort="Devos, David" uniqKey="Devos D" first="David" last="Devos">David Devos</name></author><author><name sortKey="Freeman, Leorah" sort="Freeman, Leorah" uniqKey="Freeman L" first="Leorah" last="Freeman">Leorah Freeman</name></author><author><name sortKey="Le Ber, Isabelle" sort="Le Ber, Isabelle" uniqKey="Le Ber I" first="Isabelle" last="Le Ber">Isabelle Le Ber</name></author><author><name sortKey="N Guyen, Karine" sort="N Guyen, Karine" uniqKey="N Guyen K" first="Karine" last="N'Guyen">Karine N'Guyen</name></author><author><name sortKey="Tchikviladze, Maya" sort="Tchikviladze, Maya" uniqKey="Tchikviladze M" first="Maya" last="Tchikviladzé">Maya Tchikviladzé</name></author><author><name sortKey="Labauge, Pierre" sort="Labauge, Pierre" uniqKey="Labauge P" first="Pierre" last="Labauge">Pierre Labauge</name></author><author><name sortKey="Hannequin, Didier" sort="Hannequin, Didier" uniqKey="Hannequin D" first="Didier" last="Hannequin">Didier Hannequin</name></author><author><name sortKey="Brice, Alexis" sort="Brice, Alexis" uniqKey="Brice A" first="Alexis" last="Brice">Alexis Brice</name></author><author><name sortKey="Durr, Alexandra" sort="Durr, Alexandra" uniqKey="Durr A" first="Alexandra" last="Durr">Alexandra Durr</name></author><author><name sortKey="Du Montcel, Sophie Tezenas" sort="Du Montcel, Sophie Tezenas" uniqKey="Du Montcel S" first="Sophie Tezenas" last="Du Montcel">Sophie Tezenas Du Montcel</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>Adult</term><term>Analysis of Variance</term><term>Cerebellum (pathology)</term><term>Cerebellum (physiopathology)</term><term>Disability Evaluation</term><term>Disease Progression</term><term>Female</term><term>Follow-Up Studies</term><term>Humans</term><term>Male</term><term>Middle Aged</term><term>Sample Size</term><term>Severity of Illness Index</term><term>Spinocerebellar Ataxias (diagnosis)</term><term>Spinocerebellar Ataxias (physiopathology)</term><term>Statistics as Topic</term><term>Young Adult</term></keywords><keywords scheme="MESH" qualifier="diagnosis" xml:lang="en"><term>Spinocerebellar Ataxias</term></keywords><keywords scheme="MESH" qualifier="pathology" xml:lang="en"><term>Cerebellum</term></keywords><keywords scheme="MESH" qualifier="physiopathology" xml:lang="en"><term>Cerebellum</term><term>Spinocerebellar Ataxias</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adult</term><term>Analysis of Variance</term><term>Disability Evaluation</term><term>Disease Progression</term><term>Female</term><term>Follow-Up Studies</term><term>Humans</term><term>Male</term><term>Middle Aged</term><term>Sample Size</term><term>Severity of Illness Index</term><term>Statistics as Topic</term><term>Young Adult</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Responsive ataxia rating scales are essential for determining outcome measures in clinical trials.</div></front></TEI><pubmed><MedlineCitation Owner="NLM" Status="MEDLINE"><PMID Version="1">21287600</PMID><DateCreated><Year>2011</Year><Month>04</Month><Day>04</Day></DateCreated><DateCompleted><Year>2011</Year><Month>07</Month><Day>29</Day></DateCompleted><Article PubModel="Print-Electronic"><Journal><ISSN IssnType="Electronic">1531-8257</ISSN><JournalIssue CitedMedium="Internet"><Volume>26</Volume><Issue>3</Issue><PubDate><Year>2011</Year><Month>Feb</Month><Day>15</Day></PubDate></JournalIssue><Title>Movement disorders : official journal of the Movement Disorder Society</Title><ISOAbbreviation>Mov. Disord.</ISOAbbreviation></Journal><ArticleTitle>Quantitative assessment of the evolution of cerebellar signs in spinocerebellar ataxias.</ArticleTitle><Pagination><MedlinePgn>534-8</MedlinePgn></Pagination><ELocationID EIdType="doi" ValidYN="Y">10.1002/mds.23531</ELocationID><Abstract><AbstractText Label="BACKGROUND" NlmCategory="BACKGROUND">Responsive ataxia rating scales are essential for determining outcome measures in clinical trials.</AbstractText><AbstractText Label="METHODS" NlmCategory="METHODS">We evaluated the responsiveness over time of the composite cerebellar functional severity score, a quantitative score measuring cerebellar ataxia in 133 patients with autosomal dominant cerebellar ataxias (ADCA), which were prospectively evaluated at inclusion and after one-year of follow-up. A more responsive tool was developed, the Cerebellar Functional Severity score writing, incorporating the writing test at dominant hand to the Cerebellar Functional Severity score.</AbstractText><AbstractText Label="RESULTS" NlmCategory="RESULTS">Within the one-year follow-up period, the Cerebellar Functional Severity score and its writing version increased significantly and the Scale for the Assessment and Rating of Ataxia decreased significantly reflecting increased severity of the cerebellar symptoms. The Cerebellar Functional Severity score writing responsiveness was best in genotypes SCA1, 2, and 3 compared with the other genotypes (effect size = 0.196, standardized response mean (SRM) = 0.624 versus effect size = -0.051, SRM = -0.150). The Cerebellar Functional Severity score writing used as an outcome measure would require only 163 SCA1, 2, or 3 patients per group in a two-arm interventional trial for a 50% reduction in progression and 80% of power.</AbstractText><AbstractText Label="DISCUSSION" NlmCategory="CONCLUSIONS">Our study demonstrates that the Cerebellar Functional Severity score and Cerebellar Functional Severity score writing are responsive quantitative scores for evaluating sensitivity to change in ADCA patients and can be used as outcome measures in clinical trials, especially when targeting genotypes SCA1, 2 and 3.</AbstractText><CopyrightInformation>Copyright © 2011 Movement Disorder Society.</CopyrightInformation></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Chan</LastName><ForeName>Ellis</ForeName><Initials>E</Initials><AffiliationInfo><Affiliation>Department of Biostatistics and Medical Informatics and Pitié-Salpêtrière Charles-Foix Clinical Research Unit, AP-HP, Paris, France.</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Charles</LastName><ForeName>Perrine</ForeName><Initials>P</Initials></Author><Author ValidYN="Y"><LastName>Ribai</LastName><ForeName>Pascale</ForeName><Initials>P</Initials></Author><Author ValidYN="Y"><LastName>Goizet</LastName><ForeName>Cyril</ForeName><Initials>C</Initials></Author><Author ValidYN="Y"><LastName>Marelli</LastName><ForeName>Cecilia</ForeName><Initials>C</Initials></Author><Author ValidYN="Y"><LastName>Vincitorio</LastName><ForeName>Carlo-Maria</ForeName><Initials>CM</Initials></Author><Author ValidYN="Y"><LastName>Le Bayon</LastName><ForeName>Alice</ForeName><Initials>A</Initials></Author><Author ValidYN="Y"><LastName>Guyant-Maréchal</LastName><ForeName>Lucie</ForeName><Initials>L</Initials></Author><Author ValidYN="Y"><LastName>Vandenberghe</LastName><ForeName>Nadia</ForeName><Initials>N</Initials></Author><Author ValidYN="Y"><LastName>Anheim</LastName><ForeName>Mathieu</ForeName><Initials>M</Initials></Author><Author ValidYN="Y"><LastName>Devos</LastName><ForeName>David</ForeName><Initials>D</Initials></Author><Author ValidYN="Y"><LastName>Freeman</LastName><ForeName>Leorah</ForeName><Initials>L</Initials></Author><Author ValidYN="Y"><LastName>Le Ber</LastName><ForeName>Isabelle</ForeName><Initials>I</Initials></Author><Author ValidYN="Y"><LastName>N'Guyen</LastName><ForeName>Karine</ForeName><Initials>K</Initials></Author><Author ValidYN="Y"><LastName>Tchikviladzé</LastName><ForeName>Maya</ForeName><Initials>M</Initials></Author><Author ValidYN="Y"><LastName>Labauge</LastName><ForeName>Pierre</ForeName><Initials>P</Initials></Author><Author ValidYN="Y"><LastName>Hannequin</LastName><ForeName>Didier</ForeName><Initials>D</Initials></Author><Author ValidYN="Y"><LastName>Brice</LastName><ForeName>Alexis</ForeName><Initials>A</Initials></Author><Author ValidYN="Y"><LastName>Durr</LastName><ForeName>Alexandra</ForeName><Initials>A</Initials></Author><Author ValidYN="Y"><LastName>du Montcel</LastName><ForeName>Sophie Tezenas</ForeName><Initials>ST</Initials></Author></AuthorList><Language>eng</Language><PublicationTypeList><PublicationType UI="D016428">Journal Article</PublicationType><PublicationType UI="D013485">Research Support, Non-U.S. Gov't</PublicationType></PublicationTypeList><ArticleDate DateType="Electronic"><Year>2011</Year><Month>02</Month><Day>01</Day></ArticleDate></Article><MedlineJournalInfo><Country>United States</Country><MedlineTA>Mov Disord</MedlineTA><NlmUniqueID>8610688</NlmUniqueID><ISSNLinking>0885-3185</ISSNLinking></MedlineJournalInfo><CitationSubset>IM</CitationSubset><MeshHeadingList><MeshHeading><DescriptorName MajorTopicYN="N" UI="D000328">Adult</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D000704">Analysis of Variance</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D002531">Cerebellum</DescriptorName><QualifierName MajorTopicYN="N" UI="Q000473">pathology</QualifierName><QualifierName MajorTopicYN="Y" UI="Q000503">physiopathology</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D004185">Disability Evaluation</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D018450">Disease Progression</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D005260">Female</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D005500">Follow-Up Studies</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D006801">Humans</DescriptorName></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="D018401">Sample Size</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="Y" UI="D012720">Severity of Illness Index</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D020754">Spinocerebellar Ataxias</DescriptorName><QualifierName MajorTopicYN="Y" UI="Q000175">diagnosis</QualifierName><QualifierName MajorTopicYN="Y" UI="Q000503">physiopathology</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D013223">Statistics as Topic</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D055815">Young Adult</DescriptorName></MeshHeading></MeshHeadingList></MedlineCitation><PubmedData><History><PubMedPubDate PubStatus="received"><Year>2010</Year><Month>6</Month><Day>17</Day></PubMedPubDate><PubMedPubDate PubStatus="revised"><Year>2010</Year><Month>9</Month><Day>8</Day></PubMedPubDate><PubMedPubDate PubStatus="accepted"><Year>2010</Year><Month>10</Month><Day>21</Day></PubMedPubDate><PubMedPubDate PubStatus="aheadofprint"><Year>2011</Year><Month>2</Month><Day>1</Day></PubMedPubDate><PubMedPubDate PubStatus="entrez"><Year>2011</Year><Month>2</Month><Day>3</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="pubmed"><Year>2011</Year><Month>2</Month><Day>3</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate><PubMedPubDate PubStatus="medline"><Year>2011</Year><Month>7</Month><Day>30</Day><Hour>6</Hour><Minute>0</Minute></PubMedPubDate></History><PublicationStatus>ppublish</PublicationStatus><ArticleIdList><ArticleId IdType="doi">10.1002/mds.23531</ArticleId><ArticleId IdType="pubmed">21287600</ArticleId></ArticleIdList></PubmedData></pubmed><affiliations><list><country><li>France</li></country><settlement><li>Paris</li></settlement></list><tree><noCountry><name sortKey="Anheim, Mathieu" sort="Anheim, Mathieu" uniqKey="Anheim M" first="Mathieu" last="Anheim">Mathieu Anheim</name><name sortKey="Brice, Alexis" sort="Brice, Alexis" uniqKey="Brice A" first="Alexis" last="Brice">Alexis Brice</name><name sortKey="Charles, Perrine" sort="Charles, Perrine" uniqKey="Charles P" first="Perrine" last="Charles">Perrine Charles</name><name sortKey="Devos, David" sort="Devos, David" uniqKey="Devos D" first="David" last="Devos">David Devos</name><name sortKey="Du Montcel, Sophie Tezenas" sort="Du Montcel, Sophie Tezenas" uniqKey="Du Montcel S" first="Sophie Tezenas" last="Du Montcel">Sophie Tezenas Du Montcel</name><name sortKey="Durr, Alexandra" sort="Durr, Alexandra" uniqKey="Durr A" first="Alexandra" last="Durr">Alexandra Durr</name><name sortKey="Freeman, Leorah" sort="Freeman, Leorah" uniqKey="Freeman L" first="Leorah" last="Freeman">Leorah Freeman</name><name sortKey="Goizet, Cyril" sort="Goizet, Cyril" uniqKey="Goizet C" first="Cyril" last="Goizet">Cyril Goizet</name><name sortKey="Guyant Marechal, Lucie" sort="Guyant Marechal, Lucie" uniqKey="Guyant Marechal L" first="Lucie" last="Guyant-Maréchal">Lucie Guyant-Maréchal</name><name sortKey="Hannequin, Didier" sort="Hannequin, Didier" uniqKey="Hannequin D" first="Didier" last="Hannequin">Didier Hannequin</name><name sortKey="Labauge, Pierre" sort="Labauge, Pierre" uniqKey="Labauge P" first="Pierre" last="Labauge">Pierre Labauge</name><name sortKey="Le Bayon, Alice" sort="Le Bayon, Alice" uniqKey="Le Bayon A" first="Alice" last="Le Bayon">Alice Le Bayon</name><name sortKey="Le Ber, Isabelle" sort="Le Ber, Isabelle" uniqKey="Le Ber I" first="Isabelle" last="Le Ber">Isabelle Le Ber</name><name sortKey="Marelli, Cecilia" sort="Marelli, Cecilia" uniqKey="Marelli C" first="Cecilia" last="Marelli">Cecilia Marelli</name><name sortKey="N Guyen, Karine" sort="N Guyen, Karine" uniqKey="N Guyen K" first="Karine" last="N'Guyen">Karine N'Guyen</name><name sortKey="Ribai, Pascale" sort="Ribai, Pascale" uniqKey="Ribai P" first="Pascale" last="Ribai">Pascale Ribai</name><name sortKey="Tchikviladze, Maya" sort="Tchikviladze, Maya" uniqKey="Tchikviladze M" first="Maya" last="Tchikviladzé">Maya Tchikviladzé</name><name sortKey="Vandenberghe, Nadia" sort="Vandenberghe, Nadia" uniqKey="Vandenberghe N" first="Nadia" last="Vandenberghe">Nadia Vandenberghe</name><name sortKey="Vincitorio, Carlo Maria" sort="Vincitorio, Carlo Maria" uniqKey="Vincitorio C" first="Carlo-Maria" last="Vincitorio">Carlo-Maria Vincitorio</name></noCountry><country name="France"><noRegion><name sortKey="Chan, Ellis" sort="Chan, Ellis" uniqKey="Chan E" first="Ellis" last="Chan">Ellis Chan</name></noRegion></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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