{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,8]],"date-time":"2026-10-08T04:34:32Z","timestamp":1791434072715,"version":"4.3.3"},"reference-count":36,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2022,10,23]],"date-time":"2022-10-23T00:00:00Z","timestamp":1666483200000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"publisher","award":["U01 NS104326"],"award-info":[{"award-number":["U01 NS104326"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"publisher","award":["NS104326"],"award-info":[{"award-number":["NS104326"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"publisher","award":["U01"],"award-info":[{"award-number":["U01"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["movementdisorders.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Movement Disorders"],"published-print":{"date-parts":[[2023,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>The Scale for the Assessment and Rating of Ataxia (SARA) is the reference clinical scale to assess the severity of cerebellar ataxia. In the context of upcoming therapeutic trials, a reliable clinical outcome is needed to assess the efficiency of treatments.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>The aim is to precisely assess and compare temporal dynamics of SARA and a new f\u2010SARA.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We analyzed data from four cohorts (EUROSCA, RISCA, CRC\u2010SCA, and SPATAX) comprising 1210 participants and 4092 visits. The linearity of the progression and the variability were assessed using an ordinal Bayesian mixed\u2010effect model (Leaspy). We performed sample size calculations for therapeutic trials with different scenarios to improve the responsiveness of the scale.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Seven of the eight different items had a nonlinear progression. The speed of progression was different between most of the items, with an average time for a one\u2010point increase from 3.5\u2009years [3.4; 3.6] (median, 95% credible interval) for the fastest item to 11.4 [10.9; 12.0] years. The total SARA score had a linear progression with an average time for a one\u2010point increase of 0.95 [0.92; 0.98] years. After removing the four last items and rescaling all items from 0 to 4, variability increased and progression was slower and thus would require a larger sample size in a future therapeutic trial.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>\n                      Despite a heterogeneous temporal dynamics at the item level, the global progression of SARA was linear. Changing the initial scale deteriorates the responsiveness. This new information about the temporal dynamics of the scale should help design the outcome of future clinical trials. \u00a9 2022 The Authors.\n                      <jats:italic>Movement Disorders<\/jats:italic>\n                      published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/mds.29255","type":"journal-article","created":{"date-parts":[[2022,10,23]],"date-time":"2022-10-23T12:14:25Z","timestamp":1666527265000},"page":"35-44","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":29,"title":["Temporal Dynamics of the Scale for the Assessment and Rating of Ataxia in Spinocerebellar Ataxias"],"prefix":"10.1002","volume":"38","author":[{"ORCID":"https:\/\/orcid.org\/0000-0003-3334-6035","authenticated-orcid":false,"given":"Paul","family":"Moulaire","sequence":"first","affiliation":[{"name":"Sorbonne Universit\u00e9, Paris Brain Institute, INSERM, Institut Pierre Louis d'\u00c9pid\u00e9miologie et de Sant\u00e9 Publique, INRIA, CNRS  APHP Paris 75013 France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pierre Emmanuel","family":"Poulet","sequence":"additional","affiliation":[{"name":"Sorbonne Universit\u00e9 Paris Brain Institute, INSERM, INRIA, CNRS, APHP  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emilien","family":"Petit","sequence":"additional","affiliation":[{"name":"Sorbonne Universit\u00e9 Paris Brain Institute, INSERM, INRIA, CNRS, APHP  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Thomas","family":"Klockgether","sequence":"additional","affiliation":[{"name":"German Center for Neurodegenerative Diseases (DZNE)  Bonn Germany"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexandra","family":"Durr","sequence":"additional","affiliation":[{"name":"Sorbonne Universit\u00e9 Paris Brain Institute, INSERM, INRIA, CNRS, APHP  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7180-2869","authenticated-orcid":false,"given":"Tetsuo","family":"Ashisawa","sequence":"additional","affiliation":[{"name":"Weill Cornell Medicine at the Houston Methodist Research Institute  Houston Texas USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2866-4330","authenticated-orcid":false,"given":"Sophie","family":"Tezenas du Montcel","sequence":"additional","affiliation":[{"name":"Sorbonne Universit\u00e9 Paris Brain Institute, INSERM, INRIA, CNRS, APHP  Paris France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"for the READISCA Consortium","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2022,10,23]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1474-4422(10)70183-6"},{"key":"e_1_2_8_3_1","volume-title":"StatPearls","author":"Bhandari J"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41572-019-0074-3"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1399-0004.2007.00910.x"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1002\/acn3.156"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1089\/scd.2017.0209"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1212\/01.wnl.0000219042.60538.92"},{"key":"e_1_2_8_9_1","first-page":"591","article-title":"Reliability of the Japanese version of the scale for the assessment and rating of ataxia (SARA)","volume":"61","author":"Sato K","year":"2009","journal-title":"Brain Nerve Shinkei Kenkyu No Shinpo"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1590\/S0004-282X2010000200014"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.21544"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.parkreldis.2018.12.023"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.29135"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.parkreldis.2021.08.014"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1212\/NXG.0000000000000415"},{"key":"e_1_2_8_16_1","unstructured":"Troriluzole in Adult Subjects With Spinocerebellar Ataxia \u2010 Tabular View \u2010 ClinicalTrials.gov.https:\/\/clinicaltrials.gov\/ct2\/show\/record\/NCT03701399."},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1474-4422(15)00202-1"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1186\/1750-1172-8-177"},{"key":"e_1_2_8_19_1","unstructured":"SPATAX Network: Clinical and Genetic Analysis of Spastic Paraplegia and Ataxia [Internet].c2012 [cited 2022 Oct]. 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