Gait variability as digital biomarker of disease severity in Huntington's disease.


Journal

Journal of neurology
ISSN: 1432-1459
Titre abrégé: J Neurol
Pays: Germany
ID NLM: 0423161

Informations de publication

Date de publication:
06 2020
Historique:
received: 20 09 2019
accepted: 22 01 2020
revised: 20 01 2020
pubmed: 13 2 2020
medline: 18 3 2021
entrez: 13 2 2020
Statut: ppublish

Résumé

Impaired gait plays an important role for quality of life in patients with Huntington's disease (HD). Measuring objective gait parameters in HD might provide an unbiased assessment of motor deficits in order to determine potential beneficial effects of future treatments. To objectively identify characteristic features of gait in HD patients using sensor-based gait analysis. Particularly, gait parameters were correlated to the Unified Huntington's Disease Rating Scale, total motor score (TMS), and total functional capacity (TFC). Patients with manifest HD at two German sites (n = 43) were included and clinically assessed during their annual ENROLL-HD visit. In addition, patients with HD and a cohort of age- and gender-matched controls performed a defined gait test (4 × 10 m walk). Gait patterns were recorded by inertial sensors attached to both shoes. Machine learning algorithms were applied to calculate spatio-temporal gait parameters and gait variability expressed as coefficient of variance (CV). Stride length (- 15%) and gait velocity (- 19%) were reduced, while stride (+ 7%) and stance time (+ 2%) were increased in patients with HD. However, parameters reflecting gait variability were substantially altered in HD patients (+ 17% stride length CV up to + 41% stride time CV with largest effect size) and showed strong correlations to TMS and TFC (0.416 ≤ r Sensor-based gait variability parameters were identified as clinically most relevant digital biomarker for gait impairment in HD. Altered gait variability represents characteristic irregularity of gait in HD and reflects disease severity.

Sections du résumé

BACKGROUND
Impaired gait plays an important role for quality of life in patients with Huntington's disease (HD). Measuring objective gait parameters in HD might provide an unbiased assessment of motor deficits in order to determine potential beneficial effects of future treatments.
OBJECTIVE
To objectively identify characteristic features of gait in HD patients using sensor-based gait analysis. Particularly, gait parameters were correlated to the Unified Huntington's Disease Rating Scale, total motor score (TMS), and total functional capacity (TFC).
METHODS
Patients with manifest HD at two German sites (n = 43) were included and clinically assessed during their annual ENROLL-HD visit. In addition, patients with HD and a cohort of age- and gender-matched controls performed a defined gait test (4 × 10 m walk). Gait patterns were recorded by inertial sensors attached to both shoes. Machine learning algorithms were applied to calculate spatio-temporal gait parameters and gait variability expressed as coefficient of variance (CV).
RESULTS
Stride length (- 15%) and gait velocity (- 19%) were reduced, while stride (+ 7%) and stance time (+ 2%) were increased in patients with HD. However, parameters reflecting gait variability were substantially altered in HD patients (+ 17% stride length CV up to + 41% stride time CV with largest effect size) and showed strong correlations to TMS and TFC (0.416 ≤ r
CONCLUSIONS
Sensor-based gait variability parameters were identified as clinically most relevant digital biomarker for gait impairment in HD. Altered gait variability represents characteristic irregularity of gait in HD and reflects disease severity.

Identifiants

pubmed: 32048014
doi: 10.1007/s00415-020-09725-3
pii: 10.1007/s00415-020-09725-3
pmc: PMC7293689
doi:

Substances chimiques

Biomarkers 0

Types de publication

Journal Article Multicenter Study Observational Study

Langues

eng

Sous-ensembles de citation

IM

Pagination

1594-1601

Commentaires et corrections

Type : ErratumIn

Références

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Auteurs

Heiko Gaßner (H)

Department of Molecular Neurology, University Hospital Erlangen, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Schwabachanlage 6, 91054, Erlangen, Germany.

Dennis Jensen (D)

Department of Molecular Neurology, University Hospital Erlangen, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Schwabachanlage 6, 91054, Erlangen, Germany.

F Marxreiter (F)

Department of Molecular Neurology, University Hospital Erlangen, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Schwabachanlage 6, 91054, Erlangen, Germany.

Anja Kletsch (A)

George-Huntington Institute (GHI) GmbH, Münster, Germany.

Stefan Bohlen (S)

George-Huntington Institute (GHI) GmbH, Münster, Germany.

Robin Schubert (R)

George-Huntington Institute (GHI) GmbH, Münster, Germany.

Lisa M Muratori (LM)

George-Huntington Institute (GHI) GmbH, Münster, Germany.
Rehabilitation Research and Movement Performance Laboratory (RRAMP Lab), Stony Brook University, Stony Brook, NY, USA.

Bjoern Eskofier (B)

Machine Learning and Data Analytics Lab, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Erlangen, Germany.

Jochen Klucken (J)

Department of Molecular Neurology, University Hospital Erlangen, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Schwabachanlage 6, 91054, Erlangen, Germany.
Medical Valley-Digital Health Application Center GmbH, Bamberg, Germany.
Fraunhofer Institute for Integrated Circuits IIS, Erlangen, Germany.

Jürgen Winkler (J)

Department of Molecular Neurology, University Hospital Erlangen, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Schwabachanlage 6, 91054, Erlangen, Germany.

Ralf Reilmann (R)

George-Huntington Institute (GHI) GmbH, Münster, Germany.
Department of Radiology, University of Muenster, Muenster, Germany.
Department of Neurodegenerative Diseases and Hertie-Institute for Clinical Brain Research, University of Tuebingen, Tuebingen, Germany.

Zacharias Kohl (Z)

Department of Molecular Neurology, University Hospital Erlangen, Friedrich-Alexander University Erlangen-Nürnberg (FAU), Schwabachanlage 6, 91054, Erlangen, Germany. Zacharias.Kohl@klinik.uni-regensburg.de.
Center for Rare Diseases Erlangen, University Hospital Erlangen, Erlangen, Germany. Zacharias.Kohl@klinik.uni-regensburg.de.
Department of Neurology, University of Regensburg, Regensburg, Germany. Zacharias.Kohl@klinik.uni-regensburg.de.

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