Quantitative evaluation of upper limb ataxia in spinocerebellar ataxias

Abstract Objective To quantitatively evaluate upper limb ataxia using a novel pen‐like sensor device in patients with spinocerebellar ataxia (SCA) and to assess its validity, reliability, and sensitivity to disease progression. Methods We designed a cross‐sectional and longitudinal study of patients...

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Main Authors: Yoshiyuki Kishimoto, Atsushi Hashizume, Yuta Imai, Masahiro Nakatochi, Shinichiro Yamada, Daisuke Ito, Ryota Torii, Yoshitaka Nagano, Hideo Fujimoto, Masahisa Katsuno
Format: Article
Language:English
Published: Wiley 2022-04-01
Series:Annals of Clinical and Translational Neurology
Online Access:https://doi.org/10.1002/acn3.51528
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author Yoshiyuki Kishimoto
Atsushi Hashizume
Yuta Imai
Masahiro Nakatochi
Shinichiro Yamada
Daisuke Ito
Ryota Torii
Yoshitaka Nagano
Hideo Fujimoto
Masahisa Katsuno
author_facet Yoshiyuki Kishimoto
Atsushi Hashizume
Yuta Imai
Masahiro Nakatochi
Shinichiro Yamada
Daisuke Ito
Ryota Torii
Yoshitaka Nagano
Hideo Fujimoto
Masahisa Katsuno
author_sort Yoshiyuki Kishimoto
collection DOAJ
description Abstract Objective To quantitatively evaluate upper limb ataxia using a novel pen‐like sensor device in patients with spinocerebellar ataxia (SCA) and to assess its validity, reliability, and sensitivity to disease progression. Methods We designed a cross‐sectional and longitudinal study of patients with SCA and healthy controls. Upper limb ataxia was evaluated using a device that measures the three‐dimensional position every 10 msec. Participants were instructed to move a pen‐like part of the device iteratively between two buttons. We evaluated the time, length, velocity, and variation coefficient of the stroke, and calculated the distortion index using the mean squared error. The following scales were also evaluated: Scale for the Assessment and Rating of Ataxia (SARA), the International Cooperative Ataxia Rating Scale (ICARS), and the nine‐hole pegboard test. Subjects were followed 12 months after the baseline evaluation. Results A total of 42 patients with SCA and 33 healthy controls were enrolled and evaluated. For all ataxia indices measured using the device there were significant differences between healthy controls and patients with SCA. Among the ataxia indices, the distortion index showed the strongest correlation with the SARA and ICARS upper limb score (Pearson's r = 0.647 and 0.722, respectively). Test–retest reliability was high for most of the ataxia indices. In the longitudinal analysis, the distortion index showed high standardized response mean and adjusted effect size, regardless of disease severity. Interpretation Our study demonstrated that the distortion index is a reliable functional marker that is sensitive to longitudinal change in patients with SCA.
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spelling doaj.art-621b95f8fb8c4bb8be5acc63667297fd2022-12-22T03:13:40ZengWileyAnnals of Clinical and Translational Neurology2328-95032022-04-019452953910.1002/acn3.51528Quantitative evaluation of upper limb ataxia in spinocerebellar ataxiasYoshiyuki Kishimoto0Atsushi Hashizume1Yuta Imai2Masahiro Nakatochi3Shinichiro Yamada4Daisuke Ito5Ryota Torii6Yoshitaka Nagano7Hideo Fujimoto8Masahisa Katsuno9Department of Neurology Nagoya University Graduate School of Medicine Nagoya JapanDepartment of Neurology Nagoya University Graduate School of Medicine Nagoya JapanDepartment of Basic Medicinal Sciences Nagoya University Graduate School of Pharmaceutical Sciences Nagoya JapanPublic Health Informatics Unit, Department of Integrated Health Sciences Nagoya University Graduate School of Medicine Nagoya JapanDepartment of Neurology Nagoya University Graduate School of Medicine Nagoya JapanDepartment of Neurology Nagoya University Graduate School of Medicine Nagoya JapanDepartment of Neurology Nagoya University Graduate School of Medicine Nagoya JapanDepartment of Electronic Robot Engineering Aichi University of Technology Gamagori JapanDepartment of Computer Science and Engineering Nagoya Institute of Technology Graduate School of Engineering Nagoya JapanDepartment of Neurology Nagoya University Graduate School of Medicine Nagoya JapanAbstract Objective To quantitatively evaluate upper limb ataxia using a novel pen‐like sensor device in patients with spinocerebellar ataxia (SCA) and to assess its validity, reliability, and sensitivity to disease progression. Methods We designed a cross‐sectional and longitudinal study of patients with SCA and healthy controls. Upper limb ataxia was evaluated using a device that measures the three‐dimensional position every 10 msec. Participants were instructed to move a pen‐like part of the device iteratively between two buttons. We evaluated the time, length, velocity, and variation coefficient of the stroke, and calculated the distortion index using the mean squared error. The following scales were also evaluated: Scale for the Assessment and Rating of Ataxia (SARA), the International Cooperative Ataxia Rating Scale (ICARS), and the nine‐hole pegboard test. Subjects were followed 12 months after the baseline evaluation. Results A total of 42 patients with SCA and 33 healthy controls were enrolled and evaluated. For all ataxia indices measured using the device there were significant differences between healthy controls and patients with SCA. Among the ataxia indices, the distortion index showed the strongest correlation with the SARA and ICARS upper limb score (Pearson's r = 0.647 and 0.722, respectively). Test–retest reliability was high for most of the ataxia indices. In the longitudinal analysis, the distortion index showed high standardized response mean and adjusted effect size, regardless of disease severity. Interpretation Our study demonstrated that the distortion index is a reliable functional marker that is sensitive to longitudinal change in patients with SCA.https://doi.org/10.1002/acn3.51528
spellingShingle Yoshiyuki Kishimoto
Atsushi Hashizume
Yuta Imai
Masahiro Nakatochi
Shinichiro Yamada
Daisuke Ito
Ryota Torii
Yoshitaka Nagano
Hideo Fujimoto
Masahisa Katsuno
Quantitative evaluation of upper limb ataxia in spinocerebellar ataxias
Annals of Clinical and Translational Neurology
title Quantitative evaluation of upper limb ataxia in spinocerebellar ataxias
title_full Quantitative evaluation of upper limb ataxia in spinocerebellar ataxias
title_fullStr Quantitative evaluation of upper limb ataxia in spinocerebellar ataxias
title_full_unstemmed Quantitative evaluation of upper limb ataxia in spinocerebellar ataxias
title_short Quantitative evaluation of upper limb ataxia in spinocerebellar ataxias
title_sort quantitative evaluation of upper limb ataxia in spinocerebellar ataxias
url https://doi.org/10.1002/acn3.51528
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