{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,1]],"date-time":"2026-10-01T12:37:09Z","timestamp":1790858229758,"version":"4.1.0"},"reference-count":81,"publisher":"Frontiers Media SA","license":[{"start":{"date-parts":[[2022,9,8]],"date-time":"2022-09-08T00:00:00Z","timestamp":1662595200000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/100001595","name":"Dystonia Medical Research Foundation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100001595","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100000065","name":"National Institute of Neurological Disorders and Stroke","doi-asserted-by":"publisher","award":["R01NS100874 R01NS119301 K08NS121600 5P50HD103555"],"award-info":[{"award-number":["R01NS100874 R01NS119301 K08NS121600 5P50HD103555"]}],"id":[{"id":"10.13039\/100000065","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["frontierspartnerships.org"],"crossmark-restriction":true},"short-container-title":["Dystonia"],"abstract":"<jats:p>Converging evidence from structural imaging studies in patients, the function of dystonia-causing genes, and the comorbidity of neuronal and behavioral defects all suggest that pediatric-onset dystonia is a neurodevelopmental disorder. However, to fully appreciate the contribution of altered development to dystonia, a mechanistic understanding of how networks become dysfunctional is required for early-onset dystonia. One current hurdle is that many dystonia animal models are ideally suited for studying adult phenotypes, as the neurodevelopmental features can be subtle or are complicated by broad developmental deficits. Furthermore, most assays that are used to measure dystonia are not suited for developing postnatal mice. Here, we characterize the early-onset dystonia in<jats:italic>Ptf1a<\/jats:italic><jats:sup><jats:italic>Cre<\/jats:italic><\/jats:sup><jats:italic>;Vglut2<\/jats:italic><jats:sup><jats:italic>fl\/fl<\/jats:italic><\/jats:sup>mice, which is caused by the absence of neurotransmission from inferior olive neurons onto cerebellar Purkinje cells. We investigate motor control with two paradigms that examine how altered neural function impacts key neurodevelopmental milestones seen in postnatal pups (postnatal day 7\u201311). We find that<jats:italic>Ptf1a<\/jats:italic><jats:sup><jats:italic>Cre<\/jats:italic><\/jats:sup><jats:italic>;Vglut2<\/jats:italic><jats:sup><jats:italic>fl\/fl<\/jats:italic><\/jats:sup>mice have poor performance on the negative geotaxis assay and the surface righting reflex. Interestingly, we also find that<jats:italic>Ptf1a<\/jats:italic><jats:sup><jats:italic>Cre<\/jats:italic><\/jats:sup><jats:italic>;Vglut2<\/jats:italic><jats:sup><jats:italic>fl\/fl<\/jats:italic><\/jats:sup>mice make fewer ultrasonic calls when socially isolated from their nests. Ultrasonic calls are often impaired in rodent models of autism spectrum disorders, a condition that can be comorbid with dystonia. Together, we show that these assays can serve as useful quantitative tools for investigating how neural dysfunction during development influences neonatal behaviors in a dystonia mouse model. Our data implicate a shared cerebellar circuit mechanism underlying dystonia-related motor signs and social impairments in mice.<\/jats:p>","DOI":"10.3389\/dyst.2022.10494","type":"journal-article","created":{"date-parts":[[2022,9,8]],"date-time":"2022-09-08T04:11:39Z","timestamp":1662610299000},"update-policy":"https:\/\/doi.org\/10.3389\/crossmark-policy","source":"Crossref","is-referenced-by-count":25,"title":["Quantification of Behavioral Deficits in Developing Mice With Dystonic Behaviors"],"prefix":"10.3389","volume":"1","author":[{"given":"Meike E.","family":"Van Der Heijden","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jason S.","family":"Gill","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alejandro G.","family":"Rey Hipolito","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Luis E.","family":"Salazar Leon","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Roy V.","family":"Sillitoe","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1965","published-online":{"date-parts":[[2022,9,8]]},"reference":[{"key":"B1","volume-title":"Dystonia. 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