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Eleven <jats:italic>ATXN3<\/jats:italic> mutation carrier cerebrospinal fluid samples were analysed for t\u2010tau and phosphorylated tau (p\u2010tau<jats:sup>181<\/jats:sup>). A transgenic SCA3 mouse model (MJDTg) was used to measure cerebellar t\u2010tau levels.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Plasma t\u2010tau levels were higher in mutation carriers below the age of 50 compared to controls, and the Inventory of Non\u2010Ataxia Signs was associated with t\u2010tau in ataxic patients (<jats:italic>p\u00a0<\/jats:italic>=\u00a00.004). Pre\u2010ataxic carriers showed higher cerebrospinal fluid t\u2010tau and p\u2010tau<jats:sup>181<\/jats:sup> concentrations compared to ataxic patients (<jats:italic>p\u00a0<\/jats:italic>=\u00a00.025 and <jats:italic>p\u00a0<\/jats:italic>=\u00a00.014, respectively). Cerebellar t\u2010tau was elevated in MJDTg mice compared to wild\u2010type (<jats:italic>p\u00a0<\/jats:italic>=\u00a00.033) only in the early stages of the disease. GFAP and UCHL1 did not show higher levels in mutation carriers compared to controls. Plasma NfL concentrations were higher in mutation carriers compared to controls, and differences were greater for younger carriers. The Scale for the Assessment and Rating of Ataxia was the strongest predictor of NfL in ataxic patients (<jats:italic>p\u00a0<\/jats:italic>&lt;\u00a00.001).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Our results suggest that tau might be a marker of early disease stages in SCA3. NfL can discriminate mutation carriers from controls and is associated with different clinical variables. Longitudinal studies are required to confirm their potential role as biomarkers in clinical trials.<\/jats:p><\/jats:sec>","DOI":"10.1111\/ene.15373","type":"journal-article","created":{"date-parts":[[2022,4,28]],"date-time":"2022-04-28T07:46:49Z","timestamp":1651132009000},"page":"2439-2452","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":52,"title":["Tau and neurofilament light\u2010chain as fluid biomarkers in spinocerebellar ataxia type 3"],"prefix":"10.1111","volume":"29","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-0697-3271","authenticated-orcid":false,"given":"Hector","family":"Garcia\u2010Moreno","sequence":"first","affiliation":[{"name":"Ataxia Centre Department of Clinical and Movement Neurosciences UCL Queen Square Institute of Neurology University College London  London UK"},{"name":"Department of Neurogenetics National Hospital for Neurology and Neurosurgery University College London Hospitals NHS 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