{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,5]],"date-time":"2026-06-05T11:01:40Z","timestamp":1780657300643,"version":"3.54.1"},"reference-count":31,"publisher":"Oxford University Press (OUP)","issue":"10","license":[{"start":{"date-parts":[[2026,6,4]],"date-time":"2026-06-04T00:00:00Z","timestamp":1780531200000},"content-version":"vor","delay-in-days":154,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100002243","name":"National Ataxia Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100002243","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100017336","name":"Dipartimenti di Eccellenza","doi-asserted-by":"publisher","award":["232\/2016"],"award-info":[{"award-number":["232\/2016"]}],"id":[{"id":"10.13039\/100017336","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Department CIBIO Core Facilities"},{"DOI":"10.13039\/501100008530","name":"European Regional Development Fund","doi-asserted-by":"publisher","award":["2014\u20132020"],"award-info":[{"award-number":["2014\u20132020"]}],"id":[{"id":"10.13039\/501100008530","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100008530","name":"European Regional Development Fund","doi-asserted-by":"publisher","award":["2021\u20132027"],"award-info":[{"award-number":["2021\u20132027"]}],"id":[{"id":"10.13039\/501100008530","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2026,6,1]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Mutations in Transglutaminase 6 (TG6) have been linked to a genetic form of spinocerebellar ataxia, namely SCA35. In recent years, several mutations associated with this disease have been identified. While some of them did not alter TG6 enzymatic activity, others induced a dominant-negative loss-of-function and altered subcellular localization. We previously observed that mutations identified in patients, which showed detrimental effects on neuronal viability in vitro, including mislocalization and activation of the unfolded protein response, were consistently characterized by a loss of TG6 enzymatic function. To investigate this effect in vivo, we re-derived Tgm6 knockout mice from the EMMA repository and performed behavioral characterization. We measured body weight and assessed motor performance using the rotarod, elevated beam\/beam balance test, and ladder test, beginning at 1\u00a0month of age and continuing through 16\u00a0months of age. Here, we report that TG6 loss-of-function impairs motor coordination in male mice, suggesting a sex-specific function for this enzyme.<\/jats:p>","DOI":"10.1093\/hmg\/ddag037","type":"journal-article","created":{"date-parts":[[2026,5,6]],"date-time":"2026-05-06T11:41:30Z","timestamp":1778067690000},"source":"Crossref","is-referenced-by-count":0,"title":["Partial TG6 loss of function causes motor deficits in male mice"],"prefix":"10.1093","volume":"35","author":[{"given":"Luisa","family":"Donini","sequence":"first","affiliation":[{"name":"Department of Cellular, Computational and Integrative Biology (CIBIO), University of Trento , via Sommarive 9, 38123 Trento ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Linda","family":"Sartori","sequence":"additional","affiliation":[{"name":"Department of Cellular, Computational and Integrative Biology (CIBIO), University of Trento , via Sommarive 9, 38123 Trento ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anna","family":"Barbieri","sequence":"additional","affiliation":[{"name":"Department of Cellular, Computational and Integrative Biology (CIBIO), University of Trento , via Sommarive 9, 38123 Trento ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alice","family":"Migazzi","sequence":"additional","affiliation":[{"name":"Department of Cellular, Computational and Integrative Biology (CIBIO), University of Trento , via Sommarive 9, 38123 Trento ,","place":["Italy"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sergio","family":"Robbiati","sequence":"additional","affiliation":[{"name":"Department of Cellular, Computational and Integrative Biology (CIBIO), University of Trento , via Sommarive 9, 38123 Trento ,","place":["Italy"]},{"name":"Model Organism Core Facility (MOF), Department CIBIO, University of Trento , via Sommarive 9, 38123 Trento 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