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Reliability of the Charcot-Marie-Tooth functional outcome measure

Bray, P; Cornett, KMD; Estilow, T; Pareyson, D; Zuccarino, R; Skorupinska, M; Pipis, M; ... Eichinger, KJ; + view all (2020) Reliability of the Charcot-Marie-Tooth functional outcome measure. Journal of the Peripheral Nervous System , 25 (3) pp. 288-291. 10.1111/jns.12406. Green open access

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Abstract

The CMT‐FOM is a 13‐item clinical outcome assessment (COA) that measures physical ability in adults with Charcot‐Marie‐Tooth disease (CMT). Test‐retest reliability, internal consistency and convergent validity have been established for the CMT‐FOM. This current study sought to establish inter‐rater reliability. Following an in‐person training of six international clinical evaluators we recruited 10 participants with genetically diagnosed CMT1A, (aged 18‐74 years, 6 female). Participants were evaluated using the CMT‐FOM over 2 days. Participants were given at least a 3 hour rest between evaluations, and were assessed twice each day. Following the provision of training by master trainers, all 13 items of the CMT‐FOM exhibited excellent inter‐rater reliability for raw scores (ICC1,1 0.825‐0.989) and z‐scores (ICC1,1 0.762‐0.969). Reliability of the CMT‐FOM total score was excellent (ICC1,1 0.983, 95% CI 0.958‐0.995). The CMT‐FOM is a reliable COA used by clinical evaluators internationally. The next steps are to establish further validation through psychometric evaluation of the CMT‐FOM in the Accelerate Clinical Trials in CMT (ACT‐CMT) study.

Type: Article
Title: Reliability of the Charcot-Marie-Tooth functional outcome measure
Open access status: An open access version is available from UCL Discovery
DOI: 10.1111/jns.12406
Publisher version: https://doi.org/10.1111/jns.12406
Language: English
Additional information: This version is the author accepted manuscript. For information on re-use, please refer to the publisher’s terms and conditions.
Keywords: Science & Technology, Life Sciences & Biomedicine, Clinical Neurology, Neurosciences, Neurosciences & Neurology, clinical evaluation, clinical outcome assessment, functional measurement, measurement, CMT NEUROPATHY SCORE, REFERENCE VALUES
UCL classification: UCL
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Brain Sciences
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Brain Sciences > UCL Queen Square Institute of Neurology
UCL > Provost and Vice Provost Offices > School of Life and Medical Sciences > Faculty of Brain Sciences > UCL Queen Square Institute of Neurology > Department of Neuromuscular Diseases
URI: https://discovery.ucl.ac.uk/id/eprint/10112429
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