Assessment of ataxia rating scales and cerebellar functional tests : critique and recommendations

Detalhes bibliográficos
Autor(a) principal: Perez-Lloret, Santiago
Data de Publicação: 2021
Outros Autores: Warrenburg, Bart van de, Rossi, Malco, Rodríguez-Blázquez, Carmen, Zesiewicz, Theresa, Saute, Jonas Alex Morales, Dürr, Alexandra, Nishizawa, Masatoyo, Martinez-Martin, Pablo, Stebbins, Glenn T., Schrag, Anette, Skorvanek , Matej, MDS Rating Scales Review Committee
Tipo de documento: Artigo
Idioma: eng
Título da fonte: Repositório Institucional da UFRGS
Texto Completo: http://hdl.handle.net/10183/250496
Resumo: Background: We assessed the clinimetric properties of ataxia rating scales and functional tests, and made recommendations regarding their use. Methods: A systematic literature search was conducted to identify the instruments used to rate ataxia symptoms. The identified rating scales and functional ability tests were reviewed and ranked by the panel as "recommended," "suggested," or "listed" for the assessment of patients with discrete cerebellar disorders, using previously established criteria. Results: We reviewed 14 instruments (9 rating scales and 5 functional tests). "Recommended" rating scales for the assessment of symptoms severity were: for Friedreich's ataxia, the Friedreich's Ataxia Rating Scale, the International Cooperative Ataxia Rating Scale (ICARS), and the Scale for the Assessment and Rating of Ataxia (SARA); for spinocerebellar ataxias, ICARS and SARA; for ataxia telangiectasia: ICARS and SARA; for brain tumors, SARA; for congenital disorder of glycosylation-phosphomannomutase-2 deficiency, ICARS; for cerebellar symptoms in multiple sclerosis, ICARS; for cerebellar symptoms in multiple system atrophy: Unified Multiple System Atrophy Rating Scale and ICARS; and for fragile X-associated tremor ataxia syndrome, ICARS. "Recommended" functional tests were: for Friedreich's ataxia, Ataxia Functional Composite Score and Composite Cerebellar Functional Severity Score; and for spinocerebellar ataxias, Ataxia Functional Composite Score, Composite Cerebellar Functional Severity Score, and SCA Functional Index. Conclusions: We identified some "recommended" scales and functional tests for the assessment of patients with major hereditary ataxias and other cerebellar disorders. The main limitations of these instruments include the limited assessment of patients in the more severe end of the spectrum and children. Further research in these populations is warranted.
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spelling Perez-Lloret, SantiagoWarrenburg, Bart van deRossi, MalcoRodríguez-Blázquez, CarmenZesiewicz, TheresaSaute, Jonas Alex MoralesDürr, AlexandraNishizawa, MasatoyoMartinez-Martin, PabloStebbins, Glenn T.Schrag, AnetteSkorvanek , MatejMDS Rating Scales Review Committee2022-10-27T04:52:43Z20210885-3185http://hdl.handle.net/10183/250496001151973Background: We assessed the clinimetric properties of ataxia rating scales and functional tests, and made recommendations regarding their use. Methods: A systematic literature search was conducted to identify the instruments used to rate ataxia symptoms. The identified rating scales and functional ability tests were reviewed and ranked by the panel as "recommended," "suggested," or "listed" for the assessment of patients with discrete cerebellar disorders, using previously established criteria. Results: We reviewed 14 instruments (9 rating scales and 5 functional tests). "Recommended" rating scales for the assessment of symptoms severity were: for Friedreich's ataxia, the Friedreich's Ataxia Rating Scale, the International Cooperative Ataxia Rating Scale (ICARS), and the Scale for the Assessment and Rating of Ataxia (SARA); for spinocerebellar ataxias, ICARS and SARA; for ataxia telangiectasia: ICARS and SARA; for brain tumors, SARA; for congenital disorder of glycosylation-phosphomannomutase-2 deficiency, ICARS; for cerebellar symptoms in multiple sclerosis, ICARS; for cerebellar symptoms in multiple system atrophy: Unified Multiple System Atrophy Rating Scale and ICARS; and for fragile X-associated tremor ataxia syndrome, ICARS. "Recommended" functional tests were: for Friedreich's ataxia, Ataxia Functional Composite Score and Composite Cerebellar Functional Severity Score; and for spinocerebellar ataxias, Ataxia Functional Composite Score, Composite Cerebellar Functional Severity Score, and SCA Functional Index. Conclusions: We identified some "recommended" scales and functional tests for the assessment of patients with major hereditary ataxias and other cerebellar disorders. The main limitations of these instruments include the limited assessment of patients in the more severe end of the spectrum and children. Further research in these populations is warranted.application/pdfengMovement disorders. New York. Vol. 36, no. 2 (Feb. 2021), p. 283-297Ataxia de FriedreichDoenças cerebelaresAtaxias espinocerebelaresFriedreich's ataxiaAtaxiCerebellar disordersClinical trialsRating scalesSpinocerebellar ataxiaAssessment of ataxia rating scales and cerebellar functional tests : critique and recommendationsEstrangeiroinfo:eu-repo/semantics/articleinfo:eu-repo/semantics/publishedVersioninfo:eu-repo/semantics/openAccessreponame:Repositório Institucional da UFRGSinstname:Universidade Federal do Rio Grande do Sul (UFRGS)instacron:UFRGSTEXT001151973.pdf.txt001151973.pdf.txtExtracted Texttext/plain79050http://www.lume.ufrgs.br/bitstream/10183/250496/2/001151973.pdf.txt221f2afc7872ed65e8d15af0b1f652baMD52ORIGINAL001151973.pdfTexto completo (inglês)application/pdf319985http://www.lume.ufrgs.br/bitstream/10183/250496/1/001151973.pdf1fa4ad8e72a8f20842d1402855042adaMD5110183/2504962022-10-28 04:47:48.74401oai:www.lume.ufrgs.br:10183/250496Repositório de PublicaçõesPUBhttps://lume.ufrgs.br/oai/requestopendoar:2022-10-28T07:47:48Repositório Institucional da UFRGS - Universidade Federal do Rio Grande do Sul (UFRGS)false
dc.title.pt_BR.fl_str_mv Assessment of ataxia rating scales and cerebellar functional tests : critique and recommendations
title Assessment of ataxia rating scales and cerebellar functional tests : critique and recommendations
spellingShingle Assessment of ataxia rating scales and cerebellar functional tests : critique and recommendations
Perez-Lloret, Santiago
Ataxia de Friedreich
Doenças cerebelares
Ataxias espinocerebelares
Friedreich's ataxia
Ataxi
Cerebellar disorders
Clinical trials
Rating scales
Spinocerebellar ataxia
title_short Assessment of ataxia rating scales and cerebellar functional tests : critique and recommendations
title_full Assessment of ataxia rating scales and cerebellar functional tests : critique and recommendations
title_fullStr Assessment of ataxia rating scales and cerebellar functional tests : critique and recommendations
title_full_unstemmed Assessment of ataxia rating scales and cerebellar functional tests : critique and recommendations
title_sort Assessment of ataxia rating scales and cerebellar functional tests : critique and recommendations
author Perez-Lloret, Santiago
author_facet Perez-Lloret, Santiago
Warrenburg, Bart van de
Rossi, Malco
Rodríguez-Blázquez, Carmen
Zesiewicz, Theresa
Saute, Jonas Alex Morales
Dürr, Alexandra
Nishizawa, Masatoyo
Martinez-Martin, Pablo
Stebbins, Glenn T.
Schrag, Anette
Skorvanek , Matej
MDS Rating Scales Review Committee
author_role author
author2 Warrenburg, Bart van de
Rossi, Malco
Rodríguez-Blázquez, Carmen
Zesiewicz, Theresa
Saute, Jonas Alex Morales
Dürr, Alexandra
Nishizawa, Masatoyo
Martinez-Martin, Pablo
Stebbins, Glenn T.
Schrag, Anette
Skorvanek , Matej
MDS Rating Scales Review Committee
author2_role author
author
author
author
author
author
author
author
author
author
author
author
dc.contributor.author.fl_str_mv Perez-Lloret, Santiago
Warrenburg, Bart van de
Rossi, Malco
Rodríguez-Blázquez, Carmen
Zesiewicz, Theresa
Saute, Jonas Alex Morales
Dürr, Alexandra
Nishizawa, Masatoyo
Martinez-Martin, Pablo
Stebbins, Glenn T.
Schrag, Anette
Skorvanek , Matej
MDS Rating Scales Review Committee
dc.subject.por.fl_str_mv Ataxia de Friedreich
Doenças cerebelares
Ataxias espinocerebelares
topic Ataxia de Friedreich
Doenças cerebelares
Ataxias espinocerebelares
Friedreich's ataxia
Ataxi
Cerebellar disorders
Clinical trials
Rating scales
Spinocerebellar ataxia
dc.subject.eng.fl_str_mv Friedreich's ataxia
Ataxi
Cerebellar disorders
Clinical trials
Rating scales
Spinocerebellar ataxia
description Background: We assessed the clinimetric properties of ataxia rating scales and functional tests, and made recommendations regarding their use. Methods: A systematic literature search was conducted to identify the instruments used to rate ataxia symptoms. The identified rating scales and functional ability tests were reviewed and ranked by the panel as "recommended," "suggested," or "listed" for the assessment of patients with discrete cerebellar disorders, using previously established criteria. Results: We reviewed 14 instruments (9 rating scales and 5 functional tests). "Recommended" rating scales for the assessment of symptoms severity were: for Friedreich's ataxia, the Friedreich's Ataxia Rating Scale, the International Cooperative Ataxia Rating Scale (ICARS), and the Scale for the Assessment and Rating of Ataxia (SARA); for spinocerebellar ataxias, ICARS and SARA; for ataxia telangiectasia: ICARS and SARA; for brain tumors, SARA; for congenital disorder of glycosylation-phosphomannomutase-2 deficiency, ICARS; for cerebellar symptoms in multiple sclerosis, ICARS; for cerebellar symptoms in multiple system atrophy: Unified Multiple System Atrophy Rating Scale and ICARS; and for fragile X-associated tremor ataxia syndrome, ICARS. "Recommended" functional tests were: for Friedreich's ataxia, Ataxia Functional Composite Score and Composite Cerebellar Functional Severity Score; and for spinocerebellar ataxias, Ataxia Functional Composite Score, Composite Cerebellar Functional Severity Score, and SCA Functional Index. Conclusions: We identified some "recommended" scales and functional tests for the assessment of patients with major hereditary ataxias and other cerebellar disorders. The main limitations of these instruments include the limited assessment of patients in the more severe end of the spectrum and children. Further research in these populations is warranted.
publishDate 2021
dc.date.issued.fl_str_mv 2021
dc.date.accessioned.fl_str_mv 2022-10-27T04:52:43Z
dc.type.driver.fl_str_mv Estrangeiro
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dc.identifier.uri.fl_str_mv http://hdl.handle.net/10183/250496
dc.identifier.issn.pt_BR.fl_str_mv 0885-3185
dc.identifier.nrb.pt_BR.fl_str_mv 001151973
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dc.language.iso.fl_str_mv eng
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dc.relation.ispartof.pt_BR.fl_str_mv Movement disorders. New York. Vol. 36, no. 2 (Feb. 2021), p. 283-297
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