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Multicenter Study
. 2022 Nov;37(11):2295-2301.
doi: 10.1002/mds.29206. Epub 2022 Aug 31.

Digital Gait Biomarkers Allow to Capture 1-Year Longitudinal Change in Spinocerebellar Ataxia Type 3

Winfried Ilg  1   2 , Björn Müller  1 , Jennifer Faber  3   4 , Judith van Gaalen  5 , Holger Hengel  2   6 , Ina R Vogt  4 , Guido Hennes  4 , Bart van de Warrenburg  5 , Thomas Klockgether  3   4 , Ludger Schöls  2   6 , Matthis Synofzik  2   6 ; ESMI Consortium
Affiliations
Multicenter Study

Digital Gait Biomarkers Allow to Capture 1-Year Longitudinal Change in Spinocerebellar Ataxia Type 3

Winfried Ilg et al. Mov Disord. 2022 Nov.

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.

Keywords: SCA3; gait; longitudinal analysis; motor biomarker; spinocerebellar ataxia.

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References

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