Measuring the rate of progression in Friedreich ataxia: Implications for clinical trial design
Identifieur interne : 001B88 ( Main/Exploration ); précédent : 001B87; suivant : 001B89Measuring the rate of progression in Friedreich ataxia: Implications for clinical trial design
Auteurs : Lisa S. Friedman [États-Unis] ; Jennifer M. Farmer [États-Unis] ; Susan Perlman [États-Unis] ; George Wilmot [États-Unis] ; Christopher M. Gomez [États-Unis] ; Khalaf O. Bushara [États-Unis] ; Katherine D. Mathews [États-Unis] ; S. H. Subramony [États-Unis] ; Tetsuo Ashizawa [États-Unis] ; Laura J. Balcer [États-Unis] ; Robert B. Wilson [États-Unis] ; David R. Lynch [États-Unis]Source :
- Movement Disorders [ 0885-3185 ] ; 2010-03-15.
Descripteurs français
- Pascal (Inist)
- Wicri :
- topic : Neurologie.
English descriptors
- KwdEn :
- Activities of Daily Living, Adult, Aged, Ataxia, Clinical Trials as Topic, Clinical trial, DNA, Mitochondrial (genetics), Disease Progression, Double-Blind Method, Female, Friedreich Ataxia (diagnosis), Friedreich Ataxia (genetics), Friedreich Ataxia (physiopathology), Friedreich ataxia, Humans, Iron-Binding Proteins (genetics), Male, Middle Aged, Mitochondrial disorder, Nervous system diseases, Neurologic Examination, Neurology, Point Mutation (genetics), Severity of Illness Index, Speech Disorders (diagnosis), Trinucleotide, Trinucleotide Repeats (genetics), Walking, Young Adult, ataxia, clinical neurology examination, mitochondrial disorder, natural history study, trinucleotide repeat disease.
- MESH :
- chemical , genetics : DNA, Mitochondrial, Iron-Binding Proteins.
- diagnosis : Friedreich Ataxia, Speech Disorders.
- genetics : Friedreich Ataxia, Point Mutation, Trinucleotide Repeats.
- physiopathology : Friedreich Ataxia.
- Activities of Daily Living, Adult, Aged, Clinical Trials as Topic, Disease Progression, Double-Blind Method, Female, Humans, Male, Middle Aged, Neurologic Examination, Severity of Illness Index, Walking, Young Adult.
Abstract
Friedreich ataxia is an autosomal recessive neurodegenerative disorder characterized by ataxia of all four limbs, dysarthria, and arreflexia. A variety of measures are currently used to quantify disease progression, including the Friedreich Ataxia Rating Scale, examiner‐rated functional disability scales, self‐reported activities of daily living and performance measures such as the timed 25‐foot walk, 9‐hole pegboard test, PATA speech test, and low‐contrast letter acuity vision charts. This study examines the rate of disease progression over one and two years in a cohort of 236 Friedreich ataxia patients using these scales and performance measure composites. The Friedreich Ataxia Rating Scale and performance‐measure composites captured disease progression, with a greater sensitivity to change over 2 years than over 1 year. The measures differed in their sensitivity to change and in possible bias. These results help to establish norms for progression in FRDA that can be useful in measuring the long‐term success of therapeutic agents and defining sample‐size calculations for double‐blind clinical trials. © 2010 Movement Disorder Society
Url:
- https://api.istex.fr/document/A06CB388546EB8D4B4B45FDF7F94DCE8AC0A1CCF/fulltext/pdf
- http://www.ncbi.nlm.nih.gov/pmc/articles/PMC2954653
DOI: 10.1002/mds.22912
Affiliations:
- États-Unis
- Californie, Floride, Géorgie (États-Unis), Illinois, Iowa, Minnesota, Pennsylvanie, Texas, État du Mississippi
- Iowa City
- Université de l'Iowa
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Le document en format XML
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<front><div type="abstract" xml:lang="en">Friedreich ataxia is an autosomal recessive neurodegenerative disorder characterized by ataxia of all four limbs, dysarthria, and arreflexia. A variety of measures are currently used to quantify disease progression, including the Friedreich Ataxia Rating Scale, examiner‐rated functional disability scales, self‐reported activities of daily living and performance measures such as the timed 25‐foot walk, 9‐hole pegboard test, PATA speech test, and low‐contrast letter acuity vision charts. This study examines the rate of disease progression over one and two years in a cohort of 236 Friedreich ataxia patients using these scales and performance measure composites. The Friedreich Ataxia Rating Scale and performance‐measure composites captured disease progression, with a greater sensitivity to change over 2 years than over 1 year. The measures differed in their sensitivity to change and in possible bias. These results help to establish norms for progression in FRDA that can be useful in measuring the long‐term success of therapeutic agents and defining sample‐size calculations for double‐blind clinical trials. © 2010 Movement Disorder Society</div>
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<name sortKey="Lynch, David R" sort="Lynch, David R" uniqKey="Lynch D" first="David R." last="Lynch">David R. Lynch</name>
<name sortKey="Lynch, David R" sort="Lynch, David R" uniqKey="Lynch D" first="David R." last="Lynch">David R. Lynch</name>
<name sortKey="Mathews, Katherine D" sort="Mathews, Katherine D" uniqKey="Mathews K" first="Katherine D." last="Mathews">Katherine D. Mathews</name>
<name sortKey="Perlman, Susan" sort="Perlman, Susan" uniqKey="Perlman S" first="Susan" last="Perlman">Susan Perlman</name>
<name sortKey="Subramony, S H" sort="Subramony, S H" uniqKey="Subramony S" first="S. H." last="Subramony">S. H. Subramony</name>
<name sortKey="Subramony, S H" sort="Subramony, S H" uniqKey="Subramony S" first="S. H." last="Subramony">S. H. Subramony</name>
<name sortKey="Wilmot, George" sort="Wilmot, George" uniqKey="Wilmot G" first="George" last="Wilmot">George Wilmot</name>
<name sortKey="Wilson, Robert B" sort="Wilson, Robert B" uniqKey="Wilson R" first="Robert B." last="Wilson">Robert B. Wilson</name>
</country>
</tree>
</affiliations>
</record>
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