Performance measures in Friedreich ataxia: Potential utility as clinical outcome tools
Identifieur interne : 003794 ( Main/Exploration ); précédent : 003793; suivant : 003795Performance measures in Friedreich ataxia: Potential utility as clinical outcome tools
Auteurs : David R. Lynch [États-Unis] ; Jennifer M. Farmer [États-Unis] ; Robert L. Wilson [États-Unis] ; Laura J. Balcer [États-Unis]Source :
- Movement Disorders [ 0885-3185 ] ; 2005-07.
Descripteurs français
- Pascal (Inist)
- Wicri :
- topic : Diabète.
English descriptors
- KwdEn :
- Adult, Antioxidant, Cardiomyopathy, Cross-Sectional Studies, Diabetes mellitus, Disability Evaluation, Female, Friedreich Ataxia (genetics), Friedreich Ataxia (physiopathology), Friedreich ataxia, Humans, Logistic Models, Male, Mitochondrial disorder, Nervous system diseases, Outcome Assessment (Health Care) (methods), Performance, Prognosis, Psychomotor Performance (physiology), Reproducibility of Results, Sickness Impact Profile, Trinucleotide Repeats (genetics), Visual Acuity (physiology), Walking (physiology), antioxidant, cardiomyopathy, composite measure, diabetes, mitochondrial disease.
- MESH :
- genetics : Friedreich Ataxia, Trinucleotide Repeats.
- methods : Outcome Assessment (Health Care).
- physiology : Psychomotor Performance, Visual Acuity, Walking.
- physiopathology : Friedreich Ataxia.
- Adult, Cross-Sectional Studies, Disability Evaluation, Female, Humans, Logistic Models, Male, Reproducibility of Results, Sickness Impact Profile.
Abstract
Although several neuroprotective agents have been proposed as potential therapies in Friedreich ataxia (FA), clinical trials of their efficacy are limited by a lack of sensitive outcome measures. We assessed whether performance measures (nine‐hole peg test, the timed 25‐foot walk, and low‐contrast letter acuity) provide valid measures of disease status in FA. Scores for each measure correlated significantly with neurologic disability and disease duration. Rank correlations between scores for performance measures were moderate in magnitude, suggesting that the each test captures separate yet related dimensions of neurological function in FA. Linear regression models demonstrated that scores from the nine‐hole peg test and the timed 25‐foot walk (after reciprocal transformation) were predicted by age and triplet repeat length in patients with FA. In addition, comparison of the temporal courses of change for each performance measure demonstrated that scores from the timed 25‐foot walk change early in the course of FA, nine‐hole peg test scores change slowly over the full course of the disorder, and low‐contrast letter acuity scores change in the later stages of the disease. Thus, a composite scale derived from these performance measures may provide the best overall measure for assessing disease progression throughout the illness. © 2005 Movement Disorder Society
Url:
DOI: 10.1002/mds.20449
Affiliations:
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Lynch</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Neurology, University of Pennsylvania School of Medicine, Philadelphia, Pennsylvania</wicri:regionArea><placeName><region type="state">Pennsylvanie</region></placeName></affiliation><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Pediatrics, University of Pennsylvania School of Medicine, and Children's Hospital of Philadelphia, Philadelphia, Pennsylvania</wicri:regionArea><placeName><region type="state">Pennsylvanie</region></placeName></affiliation></author><author><name sortKey="Farmer, Jennifer M" sort="Farmer, Jennifer M" uniqKey="Farmer J" first="Jennifer M." last="Farmer">Jennifer M. Farmer</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Pathology and Laboratory Medicine, University of Pennsylvania School of Medicine, Philadelphia, Pennsylvania</wicri:regionArea><placeName><region type="state">Pennsylvanie</region></placeName></affiliation></author><author><name sortKey="Wilson, Robert L" sort="Wilson, Robert L" uniqKey="Wilson R" first="Robert L." last="Wilson">Robert L. Wilson</name><affiliation wicri:level="2"><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Pathology and Laboratory Medicine, University of Pennsylvania School of Medicine, Philadelphia, Pennsylvania</wicri:regionArea><placeName><region type="state">Pennsylvanie</region></placeName></affiliation></author><author><name sortKey="Balcer, Laura J" sort="Balcer, Laura J" uniqKey="Balcer L" first="Laura J." last="Balcer">Laura J. 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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="2005-07">2005-07</date><biblScope unit="vol">20</biblScope><biblScope unit="issue">7</biblScope><biblScope unit="page" from="777">777</biblScope><biblScope unit="page" to="782">782</biblScope></imprint><idno type="ISSN">0885-3185</idno></series><idno type="istex">56C9C7F682A11ADFD80C1BD7B809FC29973612E2</idno><idno type="DOI">10.1002/mds.20449</idno><idno type="ArticleID">MDS20449</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adult</term><term>Antioxidant</term><term>Cardiomyopathy</term><term>Cross-Sectional Studies</term><term>Diabetes mellitus</term><term>Disability Evaluation</term><term>Female</term><term>Friedreich Ataxia (genetics)</term><term>Friedreich Ataxia (physiopathology)</term><term>Friedreich ataxia</term><term>Humans</term><term>Logistic Models</term><term>Male</term><term>Mitochondrial disorder</term><term>Nervous system diseases</term><term>Outcome Assessment (Health Care) (methods)</term><term>Performance</term><term>Prognosis</term><term>Psychomotor Performance (physiology)</term><term>Reproducibility of Results</term><term>Sickness Impact Profile</term><term>Trinucleotide Repeats (genetics)</term><term>Visual Acuity (physiology)</term><term>Walking (physiology)</term><term>antioxidant</term><term>cardiomyopathy</term><term>composite measure</term><term>diabetes</term><term>mitochondrial disease</term></keywords><keywords scheme="MESH" qualifier="genetics" xml:lang="en"><term>Friedreich Ataxia</term><term>Trinucleotide Repeats</term></keywords><keywords scheme="MESH" qualifier="methods" xml:lang="en"><term>Outcome Assessment (Health Care)</term></keywords><keywords scheme="MESH" qualifier="physiology" xml:lang="en"><term>Psychomotor Performance</term><term>Visual Acuity</term><term>Walking</term></keywords><keywords scheme="MESH" qualifier="physiopathology" xml:lang="en"><term>Friedreich Ataxia</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adult</term><term>Cross-Sectional Studies</term><term>Disability Evaluation</term><term>Female</term><term>Humans</term><term>Logistic Models</term><term>Male</term><term>Reproducibility of Results</term><term>Sickness Impact Profile</term></keywords><keywords scheme="Pascal" xml:lang="fr"><term>Antioxydant</term><term>Cardiomyopathie</term><term>Cytopathie mitochondriale</term><term>Diabète</term><term>Hérédodégénérescence spinocérébelleuse Friedreich</term><term>Performance</term><term>Pronostic</term><term>Système nerveux pathologie</term></keywords><keywords scheme="Wicri" type="topic" xml:lang="fr"><term>Diabète</term></keywords></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">Although several neuroprotective agents have been proposed as potential therapies in Friedreich ataxia (FA), clinical trials of their efficacy are limited by a lack of sensitive outcome measures. We assessed whether performance measures (nine‐hole peg test, the timed 25‐foot walk, and low‐contrast letter acuity) provide valid measures of disease status in FA. Scores for each measure correlated significantly with neurologic disability and disease duration. Rank correlations between scores for performance measures were moderate in magnitude, suggesting that the each test captures separate yet related dimensions of neurological function in FA. Linear regression models demonstrated that scores from the nine‐hole peg test and the timed 25‐foot walk (after reciprocal transformation) were predicted by age and triplet repeat length in patients with FA. In addition, comparison of the temporal courses of change for each performance measure demonstrated that scores from the timed 25‐foot walk change early in the course of FA, nine‐hole peg test scores change slowly over the full course of the disorder, and low‐contrast letter acuity scores change in the later stages of the disease. Thus, a composite scale derived from these performance measures may provide the best overall measure for assessing disease progression throughout the illness. © 2005 Movement Disorder Society</div></front></TEI><affiliations><list><country><li>États-Unis</li></country><region><li>Pennsylvanie</li></region></list><tree><country name="États-Unis"><region name="Pennsylvanie"><name sortKey="Lynch, David R" sort="Lynch, David R" uniqKey="Lynch D" first="David R." last="Lynch">David R. Lynch</name></region><name sortKey="Balcer, Laura J" sort="Balcer, Laura J" uniqKey="Balcer L" first="Laura J." last="Balcer">Laura J. Balcer</name><name sortKey="Farmer, Jennifer M" sort="Farmer, Jennifer M" uniqKey="Farmer J" first="Jennifer M." last="Farmer">Jennifer M. Farmer</name><name sortKey="Lynch, David R" sort="Lynch, David R" uniqKey="Lynch D" first="David R." last="Lynch">David R. Lynch</name><name sortKey="Wilson, Robert L" sort="Wilson, Robert L" uniqKey="Wilson R" first="Robert L." last="Wilson">Robert L. Wilson</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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