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Markey Charitable Trust and Beckman Institute (UIUC)"},{"name":"Department of Neurology (UAB)"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Movement Disorders"],"published-print":{"date-parts":[[2016,11]]},"abstract":"<jats:title>ABSTRACT<\/jats:title><jats:sec><jats:title>Introduction<\/jats:title><jats:p>DYT1 dystonia is an autosomal\u2010dominant movement disorder characterized by abnormal, often repetitive, movements and postures. Its hallmark feature is sustained or intermittent contractions of muscles involving co\u2010contractions of antagonist muscle pairs. The symptoms are relieved with the anticholinergic drug trihexyphenidyl. The primary mutation is a trinucleotide deletion (\u0394GAG) in <jats:italic>DYT1\/TOR1A<\/jats:italic>, which codes for torsinA. Previous studies showed that (1) heterozygous <jats:italic>Dyt1<\/jats:italic> \u0394GAG knock\u2010in mice, which have an analogous mutation in the endogenous gene, exhibit motor deficits and altered corticostriatal synaptic plasticity in the brain and (2) these deficits can be rescued by trihexyphenidyl. However, brain imaging studies suggest that the <jats:italic>Dyt1<\/jats:italic> knock\u2010in mouse models nonmanifesting mutation carriers of DYT1 dystonia. The aim of this work was to examine the hallmark features of DYT1 dystonia in the <jats:italic>Dyt1<\/jats:italic> knock\u2010in mice by analyzing muscular activities.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Wireless telemetry devices with biopotential channels were implanted to the bicep and the rectus femori muscles in <jats:italic>Dyt1<\/jats:italic> knock\u2010in mice, and muscular activities were recorded before and after trihexyphenidyl administration.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>(1) Consistent with DYT1 dystonia patients, <jats:italic>Dyt1<\/jats:italic> knock\u2010in mice showed sustained contractions and co\u2010contractions of the antagonistic bicep femoris and rectus femoris. (2) The abnormal muscle contractions were normalized by trihexyphenidyl.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>The results suggest that the motor deficits in <jats:italic>Dyt1<\/jats:italic> knock\u2010in mice are likely produced by abnormal muscle contractions, and <jats:italic>Dyt1<\/jats:italic> knock\u2010in mice can potentially be used as a manifesting disease model to study pathophysiology and develop novel therapeutics. \u00a9 2016 International Parkinson and Movement Disorder Society<\/jats:p><\/jats:sec>","DOI":"10.1002\/mds.26677","type":"journal-article","created":{"date-parts":[[2016,5,31]],"date-time":"2016-05-31T03:26:50Z","timestamp":1464665210000},"page":"1633-1639","source":"Crossref","is-referenced-by-count":24,"title":["Electromyographic evidence in support of a knock\u2010in mouse model of DYT1 Dystonia"],"prefix":"10.1002","volume":"31","author":[{"given":"Mark P.","family":"DeAndrade","sequence":"first","affiliation":[{"name":"Department of Neurology, College of Medicine,, College of Medicine University of Florida Gainesville Florida USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Amy","family":"Trongnetrpunya","sequence":"additional","affiliation":[{"name":"J. Crayton Pruitt Family Department of Biomedical Engineering, Herbert Wertheim College of Engineering University of Florida Gainesville Florida USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Fumiaki","family":"Yokoi","sequence":"additional","affiliation":[{"name":"Department of Neurology, College of Medicine,, College of Medicine University of Florida Gainesville Florida USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Chad C.","family":"Cheetham","sequence":"additional","affiliation":[{"name":"Center for Neurodegeneration and Experimental Therapeutics, Department of Neurology, School of Medicine University of Alabama at Birmingham Birmingham Alabama USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ning","family":"Peng","sequence":"additional","affiliation":[{"name":"Department of Cell, Developmental, and Integrative Biology, School of Medicine University of Alabama at Birmingham Birmingham Alabama USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"J. 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