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Dystonia is a poorly understood neurological movement disorder, often presenting in an idiopathic, isolated form across different parts of the body. The neurophysiological profile of isolated dystonia compared to healthy adults remains unclear. We conducted a systematic review with meta\u2010analysis of neurophysiologic TMS measures in people with isolated dystonia to provide a synthesized understanding of cortical neurophysiology associated with isolated dystonia. We performed a systematic database search and data were extracted independently by the two authors. Separate meta\u2010analyses were performed for TMS measures of: motor threshold, corticomotor excitability, short interval intracortical inhibition, cortical silent period, intracortical facilitation and afferent\u2010induced inhibition. Standardized mean differences were calculated using a random effects model to determine overall effect sizes and confidence intervals. Heterogeneity was explored using dystonia type subgroup analysis. The search resulted in 78 studies meeting inclusion criteria, of these 57 studies reported data in participants with focal hand dystonia, cervical dystonia, blepharospasm or spasmodic dysphonia, and were included in at least one meta\u2010analysis. The cortical silent period, short\u2010interval intracortical inhibition and afferent\u2010induced inhibition was found to be reduced in isolated dystonia compared to controls. Reduced GABAergic\u2010mediated inhibition in the primary motor cortex in idiopathic isolated dystonia's suggest interventions targeted to aberrant cortical disinhibition could provide a novel treatment. Future meta\u2010analyses require neurophysiology studies to use homogeneous cohorts of isolated dystonia participants, publish raw data values, and record electromyographic responses from dystonic musculature where possible.<\/jats:p>","DOI":"10.1111\/ejn.14987","type":"journal-article","created":{"date-parts":[[2020,9,29]],"date-time":"2020-09-29T17:50:12Z","timestamp":1601401812000},"page":"1300-1323","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["Cortical neurophysiology of primary isolated dystonia and non\u2010dystonic adults: A meta\u2010analysis"],"prefix":"10.1111","volume":"53","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-6822-4199","authenticated-orcid":false,"given":"Alana B.","family":"McCambridge","sequence":"first","affiliation":[{"name":"Graduate School of Health Discipline of Physiotherapy University of Technology Sydney Sydney NSW Australia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3067-0326","authenticated-orcid":false,"given":"Lynley V.","family":"Bradnam","sequence":"additional","affiliation":[{"name":"Department of Exercise Sciences Faculty of Science University of Auckland Auckland New Zealand"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2020,10,27]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.25475"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1097\/01.wnf.0000152044.43822.42"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.25888"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1097\/00002060-200007000-00007"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.neuropsychologia.2015.07.008"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(85)92413-4"},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.21219"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.3564-08.2008"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.brs.2009.03.004"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1152\/japplphysiol.91580.2008"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1460-9568.2009.06710.x"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.expneurol.2012.11.003"},{"key":"e_1_2_10_14_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.clinph.2016.09.008"},{"key":"e_1_2_10_15_1","doi-asserted-by":"publisher","DOI":"10.1002\/9780470743386"},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/jrsm.12"},{"key":"e_1_2_10_17_1","first-page":"321","article-title":"Writer's cramp: Cortical excitability in tasks involving proximo\u2010distal coordination","volume":"203","author":"Boyadjian A.","year":"2011","journal-title":"Acta Psychologica"},{"key":"e_1_2_10_18_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.3544-13.2013"},{"key":"e_1_2_10_19_1","doi-asserted-by":"publisher","DOI":"10.3389\/fnhum.2015.00286"},{"key":"e_1_2_10_20_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00221-008-1572-9"},{"key":"e_1_2_10_21_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.20367"},{"key":"e_1_2_10_22_1","doi-asserted-by":"publisher","DOI":"10.1002\/1097-4598(2000)999:9<::AID-MUS6>3.0.CO;2-I"},{"key":"e_1_2_10_23_1","doi-asserted-by":"publisher","DOI":"10.1007\/s002210050938"},{"key":"e_1_2_10_24_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.49.4.1054"},{"key":"e_1_2_10_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.clinph.2012.05.003"},{"key":"e_1_2_10_26_1","volume-title":"Statistical power analysis for the behavioral sciences","author":"Cohen J.","year":"1998"},{"key":"e_1_2_10_27_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awz112"},{"key":"e_1_2_10_28_1","doi-asserted-by":"publisher","DOI":"10.3390\/brainsci9040079"},{"key":"e_1_2_10_29_1","doi-asserted-by":"publisher","DOI":"10.1007\/PL00005641"},{"key":"e_1_2_10_30_1","doi-asserted-by":"publisher","DOI":"10.1007\/s002210000543"},{"key":"e_1_2_10_31_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.clinph.2007.07.005"},{"key":"e_1_2_10_32_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.27470"},{"key":"e_1_2_10_33_1","doi-asserted-by":"publisher","DOI":"10.1113\/JP275813"},{"key":"e_1_2_10_34_1","doi-asserted-by":"publisher","DOI":"10.1007\/s12311-013-0522-7"},{"key":"e_1_2_10_35_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awg188"},{"key":"e_1_2_10_36_1","first-page":"29","article-title":"Meta\u2010analysis in medical research","volume":"14","author":"Haidich A. 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