ArticleMovement disorders : official journal of the Movement Disorder Society2022
Differential Temporal Dynamics of Axial and Appendicular Ataxia in SCA3.
Article in Movement disorders : official journal of the Movement Disorder Society, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 14 papers.
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Who cites it
14 citing papers in PubMed, 23 citations in OpenAlex.
- Remote Assessment of Ataxia Severity in SCA3 Across Multiple Centers and Time Points.Annals of clinical and translational neurology · 2026Observational
- Minimal Clinically Important Difference of the Scale for the Assessment and Rating of Ataxia.Movement disorders clinical practice · 2026Article
- Digital Outcomes of Upper Limb Ataxia Capture Meaningful Longitudinal Change and Treatment Response.Movement disorders : official journal of the Movement Disorder Society · 2025Article
- Blood DDIT4 and TRIM13 Transcript Levels Mark the Early Stages of Machado-Joseph Disease.Annals of neurology · 2025Article
- Longitudinal Changes in Patient- and Clinical-Reported Outcomes in Early Spinocerebellar Ataxia Types 1, 2, 3, and 6 from the IDEA Study.Movement disorders clinical practice · 2025Article
- Psychometric Validation of the Modified Functional Scale for the Assessment and Rating of Ataxia (f-SARA) in Patients With Spinocerebellar Ataxia.Cerebellum (London, England) · 2024Article
- Content Validity of the Modified Functional Scale for the Assessment and Rating of Ataxia (f-SARA) Instrument in Spinocerebellar Ataxia.Cerebellum (London, England) · 2024Article
- Item performance of the scale for the assessment and rating of ataxia in rare and ultra-rare genetic ataxias.CPT: pharmacometrics & systems pharmacology · 2024Article
- Consensus Recommendations for Clinical Outcome Assessments and Registry Development in Ataxias: Ataxia Global Initiative (AGI) Working Group Expert Guidance.Cerebellum (London, England) · 2024Review
- Multifeature quantitative motor assessment of upper limb ataxia including drawing and reaching.Annals of clinical and translational neurology · 2024Article
- Prediction of Individual Disease Progression Including Parameter Uncertainty in Rare Neurodegenerative Diseases: The Example of Autosomal-Recessive Spastic Ataxia Charlevoix Saguenay (ARSACS).The AAPS journal · 2024Article
- Longitudinal changes of SARA scale in Friedreich ataxia: Strong influence of baseline score and age at onset.Annals of clinical and translational neurology · 2023Article
- The Homogeneous Azorean Machado-Joseph Disease Cohort: Characterization and Contributions to Advances in Research.Biomedicines · 2023Review
- Temporal Dynamics of the Scale for the Assessment and Rating of Ataxia in Spinocerebellar Ataxias.Movement disorders : official journal of the Movement Disorder Society · 2023Article
Corrections and comments
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Authors and funding
23 authors at 15 institutions in 6 countries.
Funding
Abstract
backgroundDisease severity in spinocerebellar ataxia type 3 (SCA3) is commonly defined by the Scale for the Assessment and Rating of Ataxia (SARA) sum score, but little is known about the contributions and progression patterns of individual items.
objectivesTo investigate the temporal dynamics of SARA item scores in SCA3 patients and evaluate if clinical and demographic factors are differentially associated with evolution of axial and appendicular ataxia.
methodsIn a prospective, multinational cohort study involving 11 European and 2 US sites, SARA scores were determined longitudinally in 223 SCA3 patients with a follow-up assessment after 1 year.
resultsAn increase in SARA score from 10 to 20 points was mainly driven by axial and speech items, with a markedly smaller contribution of appendicular items. Finger chase and nose-finger test scores not only showed the lowest variability at baseline, but also the least deterioration at follow-up. Compared with the full set of SARA items, omission of both tests would result in lower sample size requirements for therapeutic trials. Sex was associated with change in SARA sum score and appendicular, but not axial, subscore, with a significantly faster progression in men. Despite considerable interindividual variability, the average annual progression rate of SARA score was approximately three times higher in subjects with a disease duration over 10 years than in those within 10 years from onset.
conclusionOur findings provide evidence for a difference in temporal dynamics between axial and appendicular ataxia in SCA3 patients, which will help inform the design of clinical trials and development of new (etiology-specific) outcome measures. © 2022 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.
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