Journal Article

DZNE-2022-01483

http://join2-wiki.gsi.de/foswiki/pub/Main/Artwork/join2_logo100x88.png Digital Gait Biomarkers Allow to Capture 1-Year Longitudinal Change in Spinocerebellar Ataxia Type 3.

Ilg, W. (First author)DZNE* ; Müller, B. ; Faber, J.DZNE* ; van Gaalen, J. ; Hengel, H.DZNE* ; Vogt, I. R.DZNE* ; Hennes, G.DZNE* ; van de Warrenburg, B. ; Klockgether, T.DZNE* ; Schöls, L.DZNE* ; Synofzik, M. (Last author)DZNE* ; Consortium, E. (Collaboration Author)

2022
Wiley New York, NY

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Please use a persistent id in citations: doi:10.1002/mds.29206

Abstract: Measures of step variability and body sway during gait have shown to correlate with clinical ataxia severity in several cross-sectional studies. However, to serve as a valid progression biomarker, these gait measures have to prove their sensitivity to robustly capture longitudinal change, ideally within short time frames (eg, 1 year). We present the first multicenter longitudinal gait analysis study in spinocerebellar ataxias. We performed a combined cross-sectional (n = 28) and longitudinal (1-year interval, n = 17) analysis in Spinocerebellar Ataxia type 3 subjects (including seven preataxic mutation carriers). Longitudinal analysis showed significant change in gait measures between baseline and 1-year follow-up, with high effect sizes (stride length variability: P = 0.01, effect size rprb = 0.66; lateral sway: P = 0.007, rprb = 0.73). Sample size estimation for lateral sway indicates a required cohort size of n = 43 for detecting a 50% reduction of natural progression, compared with n = 240 for the clinical ataxia score Scale for the Assessment and Rating of Ataxia (SARA). These measures thus present promising motor biomarkers for upcoming interventional studies. © 2022 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.

Keyword(s): Humans (MeSH) ; Machado-Joseph Disease: diagnosis (MeSH) ; Cross-Sectional Studies (MeSH) ; Disease Progression (MeSH) ; Gait (MeSH) ; Cerebellar Ataxia (MeSH) ; Ataxia (MeSH) ; Biomarkers (MeSH) ; SCA3 ; gait ; longitudinal analysis ; motor biomarker ; spinocerebellar ataxia

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Contributing Institute(s):

1. Parkinson Genetics (AG Gasser)
2. Patient Studies Bonn (Patient Studies Bonn)
3. Patient Studies (AG Klockgether)

Research Program(s):

1. 353 - Clinical and Health Care Research (POF4-353) (POF4-353)

Appears in the scientific report 2022

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Database coverage:
; ; ; Clarivate Analytics Master Journal List ; Current Contents - Clinical Medicine ; Current Contents - Life Sciences ; DEAL Wiley ; Ebsco Academic Search ; Essential Science Indicators ; IF >= 5 ; JCR ; Nationallizenz ; SCOPUS ; Science Citation Index Expanded ; Web of Science Core Collection

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