{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,3]],"date-time":"2026-10-03T21:25:16Z","timestamp":1791062716802,"version":"4.1.0"},"reference-count":22,"publisher":"Wiley","issue":"12","license":[{"start":{"date-parts":[[2025,9,4]],"date-time":"2025-09-04T00:00:00Z","timestamp":1756944000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2025,9,4]],"date-time":"2025-09-04T00:00:00Z","timestamp":1756944000000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/501100001807","name":"Funda\u00e7\u00e3o de Amparo \u00e0 Pesquisa do Estado de S\u00e3o Paulo","doi-asserted-by":"publisher","award":["#2013\/07559\u20103"],"award-info":[{"award-number":["#2013\/07559\u20103"]}],"id":[{"id":"10.13039\/501100001807","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["movementdisorders.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Movement Disord Clin Pract"],"published-print":{"date-parts":[[2025,12]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>The progression of brain damage in CANVAS\/RFC1 remains unclear.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>To describe longitudinal brain changes in CANVAS\/RFC1.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Ten RFC1\u2010positive patients and 10 controls underwent 3T\u2010MRI scans 2\u2009years apart. We analyzed cerebral\u00a0gray and white matter using FreeSurfer and DTI multiatlas, the cerebellum with CerebNet, and the spinal cord with Spinal Cord Toolbox, respectively. Ataxia severity was measured with SARA. Longitudinal changes were assessed using mixed\u2010effect models and Standardized Response Mean (SRM).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>CANVAS\/RFC1 patients (mean disease duration: 8.5\u2009\u00b1\u20093.7\u2009years) showed progressive atrophy in the brainstem (SRM:\u20103.5), left hippocampus (SRM:\u20102.1), right thalamus (SRM:\u20102.6), left cerebellar lobules VI (SRM:\u20101.1) and X (SRM:\u20100.7), and reduced spinal cord area at C1 and C2 (SRM:\u20100.7 each). SARA variation showed lower sensitivity (SRM:0.7).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Patients with CANVAS\/RFC1 showed progressive, asymmetrical volume reduction of structures in the brain and spinal cord (C1 and C2), over a period of 2\u2009years, using 3\u2010tesla MRI.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/mdc3.70344","type":"journal-article","created":{"date-parts":[[2025,9,5]],"date-time":"2025-09-05T06:19:21Z","timestamp":1757053161000},"page":"2311-2316","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Longitudinal Evaluation of Ataxia and Brain Structural Changes in\n                    <scp>\n                      <i>RFC1<\/i>\n                    <\/scp>\n                    \u2010Related Disorder"],"prefix":"10.1002","volume":"12","author":[{"ORCID":"https:\/\/orcid.org\/0009-0007-0790-8870","authenticated-orcid":false,"given":"Camila C.","family":"Lobo","sequence":"first","affiliation":[{"name":"Department of Neurology School of Medical Sciences \u2013 University of Campinas (UNICAMP)  Campinas Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8453-0313","authenticated-orcid":false,"given":"Thiago J.R.","family":"Rezende","sequence":"additional","affiliation":[{"name":"Department of Neurology School of Medical Sciences \u2013 University of Campinas (UNICAMP)  Campinas Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Luciana R.","family":"Pimentel\u2010Silva","sequence":"additional","affiliation":[{"name":"Department of Neurology School of Medical Sciences \u2013 University of Campinas (UNICAMP)  Campinas Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0980-1114","authenticated-orcid":false,"given":"Gustavo M.","family":"Jarola","sequence":"additional","affiliation":[{"name":"Department of Neurology School of Medical Sciences \u2013 University of Campinas (UNICAMP)  Campinas Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nadson B.S.","family":"Santos","sequence":"additional","affiliation":[{"name":"Department of Neurology School of Medical Sciences \u2013 University of Campinas (UNICAMP)  Campinas Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gabriel","family":"da Silva Schmitt","sequence":"additional","affiliation":[{"name":"Department of Neurology School of Medical Sciences \u2013 University of Campinas (UNICAMP)  Campinas Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paula C.A.A.P.","family":"Matos","sequence":"additional","affiliation":[{"name":"Department of Neurology, General Neurology and Ataxia Unit Federal University of Sao Paulo (UNIFESP)  S\u00e3o Paulo Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Fabr\u00edcio D.","family":"Lima","sequence":"additional","affiliation":[{"name":"Department of Neurology School of Medical Sciences \u2013 University of Campinas (UNICAMP)  Campinas Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alberto R.M.","family":"Martinez","sequence":"additional","affiliation":[{"name":"Department of Neurology School of Medical Sciences \u2013 University of Campinas (UNICAMP)  Campinas Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Orlando G.P.","family":"Barsottini","sequence":"additional","affiliation":[{"name":"Department of Neurology, General Neurology and Ataxia Unit Federal University of Sao Paulo (UNIFESP)  S\u00e3o Paulo Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1672-8894","authenticated-orcid":false,"given":"Jos\u00e9 Luiz","family":"Pedroso","sequence":"additional","affiliation":[{"name":"Department of Neurology, General Neurology and Ataxia Unit Federal University of Sao Paulo (UNIFESP)  S\u00e3o Paulo Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"suffix":"Jr,","given":"Wilson","family":"Marques","sequence":"additional","affiliation":[{"name":"Department of Neurosciences School of Medicine \u2013 University of S\u00e3o Paulo at Ribeir\u00e3o Preto (USP\u2010RP)  Ribeir\u00e3o Preto Brazil"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0898-2419","authenticated-orcid":false,"suffix":"Jr,","given":"Marcondes C.","family":"Fran\u00e7a","sequence":"additional","affiliation":[{"name":"Department of Neurology School of Medical Sciences \u2013 University of Campinas (UNICAMP)  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