Sensorimotor adaptation as a behavioural biomarker of early spinocerebellar ataxia type 6

Abstract Early detection of the behavioural deficits of neurodegenerative diseases may help to describe the pathogenesis of such diseases and establish important biomarkers of disease progression. The aim of this study was to identify how sensorimotor adaptation of the upper limb, a cerebellar-depen...

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Autores principales:	Muriel T. N. Panouillères, Raed A. Joundi, Sonia Benitez-Rivero, Binith Cheeran, Christopher R. Butler, Andrea H. Németh, R. Chris Miall, Ned Jenkinson
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2017
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/834bdd1190104655a9bc9d8d4d367e37
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spelling	oai:doaj.org-article:834bdd1190104655a9bc9d8d4d367e372021-12-02T12:32:53ZSensorimotor adaptation as a behavioural biomarker of early spinocerebellar ataxia type 610.1038/s41598-017-02469-72045-2322https://doaj.org/article/834bdd1190104655a9bc9d8d4d367e372017-05-01T00:00:00Zhttps://doi.org/10.1038/s41598-017-02469-7https://doaj.org/toc/2045-2322Abstract Early detection of the behavioural deficits of neurodegenerative diseases may help to describe the pathogenesis of such diseases and establish important biomarkers of disease progression. The aim of this study was to identify how sensorimotor adaptation of the upper limb, a cerebellar-dependent process restoring movement accuracy after introduction of a perturbation, is affected at the pre-clinical and clinical stages of spinocerebellar ataxia type 6 (SCA6), an inherited neurodegenerative disease. We demonstrate that initial adaptation to the perturbation was significantly impaired in the eighteen individuals with clinical motor symptoms but mostly preserved in the five pre-clinical individuals. Moreover, the amount of error reduction correlated with the clinical symptoms, with the most symptomatic patients adapting the least. Finally both pre-clinical and clinical individuals showed significantly reduced de-adaptation performance after the perturbation was removed in comparison to the control participants. Thus, in this large study of motor features in SCA6, we provide novel evidence for the existence of subclinical motor dysfunction at a pre-clinical stage of SCA6. Our findings show that testing sensorimotor de-adaptation could provide a potential predictor of future motor deficits in SCA6.Muriel T. N. PanouillèresRaed A. JoundiSonia Benitez-RiveroBinith CheeranChristopher R. ButlerAndrea H. NémethR. Chris MiallNed JenkinsonNature PortfolioarticleMedicineRScienceQENScientific Reports, Vol 7, Iss 1, Pp 1-12 (2017)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Muriel T. N. Panouillères Raed A. Joundi Sonia Benitez-Rivero Binith Cheeran Christopher R. Butler Andrea H. Németh R. Chris Miall Ned Jenkinson Sensorimotor adaptation as a behavioural biomarker of early spinocerebellar ataxia type 6
description	Abstract Early detection of the behavioural deficits of neurodegenerative diseases may help to describe the pathogenesis of such diseases and establish important biomarkers of disease progression. The aim of this study was to identify how sensorimotor adaptation of the upper limb, a cerebellar-dependent process restoring movement accuracy after introduction of a perturbation, is affected at the pre-clinical and clinical stages of spinocerebellar ataxia type 6 (SCA6), an inherited neurodegenerative disease. We demonstrate that initial adaptation to the perturbation was significantly impaired in the eighteen individuals with clinical motor symptoms but mostly preserved in the five pre-clinical individuals. Moreover, the amount of error reduction correlated with the clinical symptoms, with the most symptomatic patients adapting the least. Finally both pre-clinical and clinical individuals showed significantly reduced de-adaptation performance after the perturbation was removed in comparison to the control participants. Thus, in this large study of motor features in SCA6, we provide novel evidence for the existence of subclinical motor dysfunction at a pre-clinical stage of SCA6. Our findings show that testing sensorimotor de-adaptation could provide a potential predictor of future motor deficits in SCA6.
format	article
author	Muriel T. N. Panouillères Raed A. Joundi Sonia Benitez-Rivero Binith Cheeran Christopher R. Butler Andrea H. Németh R. Chris Miall Ned Jenkinson
author_facet	Muriel T. N. Panouillères Raed A. Joundi Sonia Benitez-Rivero Binith Cheeran Christopher R. Butler Andrea H. Németh R. Chris Miall Ned Jenkinson
author_sort	Muriel T. N. Panouillères
title	Sensorimotor adaptation as a behavioural biomarker of early spinocerebellar ataxia type 6
title_short	Sensorimotor adaptation as a behavioural biomarker of early spinocerebellar ataxia type 6
title_full	Sensorimotor adaptation as a behavioural biomarker of early spinocerebellar ataxia type 6
title_fullStr	Sensorimotor adaptation as a behavioural biomarker of early spinocerebellar ataxia type 6
title_full_unstemmed	Sensorimotor adaptation as a behavioural biomarker of early spinocerebellar ataxia type 6
title_sort	sensorimotor adaptation as a behavioural biomarker of early spinocerebellar ataxia type 6
publisher	Nature Portfolio
publishDate	2017
url	https://doaj.org/article/834bdd1190104655a9bc9d8d4d367e37
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Sensorimotor adaptation as a behavioural biomarker of early spinocerebellar ataxia type 6

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