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Longitudinal multi-modal muscle-based biomarker assessment in motor neuron disease.


ABSTRACT: BACKGROUND:Clinical phenotypic heterogeneity represents a major barrier to trials in motor neuron disease (MND) and objective surrogate outcome measures are required, especially for slowly progressive patients. We assessed responsiveness of clinical, electrophysiological and radiological muscle-based assessments to detect MND-related progression. MATERIALS AND METHODS:A prospective, longitudinal cohort study of 29 MND patients and 22 healthy controls was performed. Clinical measures, electrophysiological motor unit number index/size (MUNIX/MUSIX) and relative T2- and diffusion-weighted whole-body muscle magnetic resonance (MR) were assessed three times over 12 months. Multi-variable regression models assessed between-group differences, clinico-electrophysiological associations, and longitudinal changes. Standardized response means (SRMs) assessed sensitivity to change over 12 months. RESULTS:MND patients exhibited 18% higher whole-body mean muscle relative T2-signal than controls (95% CI 7-29%, p?

SUBMITTER: Jenkins TM 

PROVIDER: S-EPMC6954906 | biostudies-literature | 2020 Jan

REPOSITORIES: biostudies-literature

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Longitudinal multi-modal muscle-based biomarker assessment in motor neuron disease.

Jenkins Thomas M TM   Alix James J P JJP   Fingret Jacob J   Esmail Taniya T   Hoggard Nigel N   Baster Kathleen K   McDermott Christopher J CJ   Wilkinson Iain D ID   Shaw Pamela J PJ  

Journal of neurology 20191017 1


<h4>Background</h4>Clinical phenotypic heterogeneity represents a major barrier to trials in motor neuron disease (MND) and objective surrogate outcome measures are required, especially for slowly progressive patients. We assessed responsiveness of clinical, electrophysiological and radiological muscle-based assessments to detect MND-related progression.<h4>Materials and methods</h4>A prospective, longitudinal cohort study of 29 MND patients and 22 healthy controls was performed. Clinical measur  ...[more]

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