Cargando…

Objectively characterizing Huntington’s disease using a novel upper limb dexterity test

BACKGROUND: The Clinch Token Transfer Test (C3t) is a bi-manual coin transfer task that incorporates cognitive tasks to add complexity. This study explored the concurrent and convergent validity of the C3t as a simple, objective assessment of impairment that is reflective of disease severity in Hunt...

Descripción completa

Detalles Bibliográficos
Autores principales: Woodgate, Samuel, Morgan-Jones, Philippa, Clinch, Susanne, Drew, Cheney, Playle, Rebecca, Bennasar, Mohamed, Hicks, Yulia, Holt, Catherine, Bachoud-Lévi, Anne-Catherine, Massart, Renaud, Craufurd, David, Kirby, Nigel, Hamana, Katy, Schubert, Robin, Reilmann, Ralf, Rosser, Anne, Busse, Monica
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Springer Berlin Heidelberg 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7868671/
https://www.ncbi.nlm.nih.gov/pubmed/33555419
http://dx.doi.org/10.1007/s00415-020-10375-8
_version_ 1783648497880268800
author Woodgate, Samuel
Morgan-Jones, Philippa
Clinch, Susanne
Drew, Cheney
Playle, Rebecca
Bennasar, Mohamed
Hicks, Yulia
Holt, Catherine
Bachoud-Lévi, Anne-Catherine
Massart, Renaud
Craufurd, David
Kirby, Nigel
Hamana, Katy
Schubert, Robin
Reilmann, Ralf
Rosser, Anne
Busse, Monica
author_facet Woodgate, Samuel
Morgan-Jones, Philippa
Clinch, Susanne
Drew, Cheney
Playle, Rebecca
Bennasar, Mohamed
Hicks, Yulia
Holt, Catherine
Bachoud-Lévi, Anne-Catherine
Massart, Renaud
Craufurd, David
Kirby, Nigel
Hamana, Katy
Schubert, Robin
Reilmann, Ralf
Rosser, Anne
Busse, Monica
author_sort Woodgate, Samuel
collection PubMed
description BACKGROUND: The Clinch Token Transfer Test (C3t) is a bi-manual coin transfer task that incorporates cognitive tasks to add complexity. This study explored the concurrent and convergent validity of the C3t as a simple, objective assessment of impairment that is reflective of disease severity in Huntington’s, that is not reliant on clinical expertise for administration. METHODS: One-hundred-and-five participants presenting with pre-manifest (n = 16) or manifest (TFC-Stage-1 n = 39; TFC-Stage-2 n = 43; TFC-Stage-3 n = 7) Huntington’s disease completed the Unified Huntington’s Disease Rating Scale and the C3t at baseline. Of these, thirty-three were followed up after 12 months. Regression was used to estimate baseline individual and composite clinical scores (including cognitive, motor, and functional ability) using baseline C3t scores. Correlations between C3t and clinical scores were assessed using Spearman’s R and visually inspected in relation to disease severity using scatterplots. Effect size over 12 months provided an indication of longitudinal behaviour of the C3t in relation to clinical measures. RESULTS: Baseline C3t scores predicted baseline clinical scores to within 9–13% accuracy, being associated with individual and composite clinical scores. Changes in C3t scores over 12 months were small ([Formula: see text] ≤ 0.15) and mirrored the change in clinical scores. CONCLUSION: The C3t demonstrates promise as a simple, easy to administer, objective outcome measure capable of predicting impairment that is reflective of Huntington’s disease severity and offers a viable solution to support remote clinical monitoring. It may also offer utility as a screening tool for recruitment to clinical trials given preliminary indications of association with the prognostic index normed for Huntington’s disease. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s00415-020-10375-8.
format Online
Article
Text
id pubmed-7868671
institution National Center for Biotechnology Information
language English
publishDate 2021
publisher Springer Berlin Heidelberg
record_format MEDLINE/PubMed
spelling pubmed-78686712021-02-09 Objectively characterizing Huntington’s disease using a novel upper limb dexterity test Woodgate, Samuel Morgan-Jones, Philippa Clinch, Susanne Drew, Cheney Playle, Rebecca Bennasar, Mohamed Hicks, Yulia Holt, Catherine Bachoud-Lévi, Anne-Catherine Massart, Renaud Craufurd, David Kirby, Nigel Hamana, Katy Schubert, Robin Reilmann, Ralf Rosser, Anne Busse, Monica J Neurol Original Communication BACKGROUND: The Clinch Token Transfer Test (C3t) is a bi-manual coin transfer task that incorporates cognitive tasks to add complexity. This study explored the concurrent and convergent validity of the C3t as a simple, objective assessment of impairment that is reflective of disease severity in Huntington’s, that is not reliant on clinical expertise for administration. METHODS: One-hundred-and-five participants presenting with pre-manifest (n = 16) or manifest (TFC-Stage-1 n = 39; TFC-Stage-2 n = 43; TFC-Stage-3 n = 7) Huntington’s disease completed the Unified Huntington’s Disease Rating Scale and the C3t at baseline. Of these, thirty-three were followed up after 12 months. Regression was used to estimate baseline individual and composite clinical scores (including cognitive, motor, and functional ability) using baseline C3t scores. Correlations between C3t and clinical scores were assessed using Spearman’s R and visually inspected in relation to disease severity using scatterplots. Effect size over 12 months provided an indication of longitudinal behaviour of the C3t in relation to clinical measures. RESULTS: Baseline C3t scores predicted baseline clinical scores to within 9–13% accuracy, being associated with individual and composite clinical scores. Changes in C3t scores over 12 months were small ([Formula: see text] ≤ 0.15) and mirrored the change in clinical scores. CONCLUSION: The C3t demonstrates promise as a simple, easy to administer, objective outcome measure capable of predicting impairment that is reflective of Huntington’s disease severity and offers a viable solution to support remote clinical monitoring. It may also offer utility as a screening tool for recruitment to clinical trials given preliminary indications of association with the prognostic index normed for Huntington’s disease. SUPPLEMENTARY INFORMATION: The online version contains supplementary material available at 10.1007/s00415-020-10375-8. Springer Berlin Heidelberg 2021-02-08 2021 /pmc/articles/PMC7868671/ /pubmed/33555419 http://dx.doi.org/10.1007/s00415-020-10375-8 Text en © The Author(s) 2021 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) .
spellingShingle Original Communication
Woodgate, Samuel
Morgan-Jones, Philippa
Clinch, Susanne
Drew, Cheney
Playle, Rebecca
Bennasar, Mohamed
Hicks, Yulia
Holt, Catherine
Bachoud-Lévi, Anne-Catherine
Massart, Renaud
Craufurd, David
Kirby, Nigel
Hamana, Katy
Schubert, Robin
Reilmann, Ralf
Rosser, Anne
Busse, Monica
Objectively characterizing Huntington’s disease using a novel upper limb dexterity test
title Objectively characterizing Huntington’s disease using a novel upper limb dexterity test
title_full Objectively characterizing Huntington’s disease using a novel upper limb dexterity test
title_fullStr Objectively characterizing Huntington’s disease using a novel upper limb dexterity test
title_full_unstemmed Objectively characterizing Huntington’s disease using a novel upper limb dexterity test
title_short Objectively characterizing Huntington’s disease using a novel upper limb dexterity test
title_sort objectively characterizing huntington’s disease using a novel upper limb dexterity test
topic Original Communication
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7868671/
https://www.ncbi.nlm.nih.gov/pubmed/33555419
http://dx.doi.org/10.1007/s00415-020-10375-8
work_keys_str_mv AT woodgatesamuel objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT morganjonesphilippa objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT clinchsusanne objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT drewcheney objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT playlerebecca objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT bennasarmohamed objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT hicksyulia objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT holtcatherine objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT bachoudleviannecatherine objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT massartrenaud objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT craufurddavid objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT kirbynigel objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT hamanakaty objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT schubertrobin objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT reilmannralf objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT rosseranne objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest
AT bussemonica objectivelycharacterizinghuntingtonsdiseaseusinganovelupperlimbdexteritytest