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We adopted an established cerebellar dystonia mouse model by injecting ouabain to examine the contribution of the cerebellum. Initially, we examined whether the entopeduncular nucleus (EPN), substantia nigra pars reticulata (SNr), globus pallidus externus (GPe) and striatal neurons were activated in the model. Next, we examined whether administration of a dopamine D1 receptor agonist and dopamine D2 receptor antagonist or selective ablation of striatal parvalbumin (PV, encoded by Pvalb)-expressing interneurons could modulate the involuntary movements of the mice. The cerebellar dystonia mice had a higher number of cells positive for c-fos (encoded by Fos) in the EPN, SNr and GPe, as well as a higher positive ratio of c-fos in striatal PV interneurons, than those in control mice. Furthermore, systemic administration of combined D1 receptor agonist and D2 receptor antagonist and selective ablation of striatal PV interneurons relieved the involuntary movements of the mice. Abnormalities in the motor loop of the basal ganglia could be crucially involved in cerebellar dystonia, and modulating PV interneurons might provide a novel treatment strategy.<\/jats:p>","DOI":"10.1242\/dmm.050338","type":"journal-article","created":{"date-parts":[[2024,4,15]],"date-time":"2024-04-15T03:55:47Z","timestamp":1713153347000},"source":"Crossref","is-referenced-by-count":7,"title":["Striatal parvalbumin interneurons are activated in a mouse model of cerebellar dystonia"],"prefix":"10.1242","volume":"17","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6206-8471","authenticated-orcid":false,"given":"Taku","family":"Matsuda","sequence":"first","affiliation":[{"name":"Graduate School of Biomedical Sciences, Tokushima University 1 Department of Neurosurgery , , Tokushima 770-8503 , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4919-6441","authenticated-orcid":false,"given":"Ryoma","family":"Morigaki","sequence":"additional","affiliation":[{"name":"Graduate School of Biomedical Sciences, Tokushima University 1 Department of Neurosurgery , , Tokushima 770-8503 , Japan"},{"name":"Graduate School of Biomedical Sciences, Tokushima University 2 Department of Advanced Brain Research , , Tokushima 770-8503 , Japan"},{"name":"Parkinson's Disease and Dystonia Research Center, Tokushima University Hospital 3 , Tokushima 770-8503 , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroaki","family":"Hayasawa","sequence":"additional","affiliation":[{"name":"Graduate School of Biomedical Sciences, Tokushima University 1 Department of Neurosurgery , , Tokushima 770-8503 , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroshi","family":"Koyama","sequence":"additional","affiliation":[{"name":"Graduate School of Biomedical Sciences, Tokushima University 1 Department of Neurosurgery , , Tokushima 770-8503 , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Teruo","family":"Oda","sequence":"additional","affiliation":[{"name":"Graduate School of Biomedical Sciences, Tokushima University 2 Department of Advanced Brain Research , , Tokushima 770-8503 , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kazuhisa","family":"Miyake","sequence":"additional","affiliation":[{"name":"Graduate School of Biomedical Sciences, Tokushima University 1 Department of Neurosurgery , , Tokushima 770-8503 , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yasushi","family":"Takagi","sequence":"additional","affiliation":[{"name":"Graduate School of Biomedical Sciences, Tokushima University 1 Department of Neurosurgery , , Tokushima 770-8503 , Japan"},{"name":"Graduate School of Biomedical Sciences, Tokushima University 2 Department of Advanced Brain Research , , Tokushima 770-8503 , Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2024,5,14]]},"reference":[{"key":"2024051410170122900_DMM050338C1","doi-asserted-by":"publisher","first-page":"604","DOI":"10.1111\/ejn.13949","article-title":"Cholinergic modulation of striatal microcircuits","volume":"49","author":"Abudukeyoumu","year":"2019","journal-title":"Eur. 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