{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,9]],"date-time":"2026-10-09T22:18:47Z","timestamp":1791584327885,"version":"4.3.4"},"reference-count":49,"publisher":"Wiley","issue":"7","license":[{"start":{"date-parts":[[2024,4,22]],"date-time":"2024-04-22T00:00:00Z","timestamp":1713744000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100003196","name":"Ministero della Salute","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100003196","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["movementdisorders.onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Movement Disorders"],"published-print":{"date-parts":[[2024,7]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>The evidence in the effectiveness of deep brain stimulation in children with medication\u2010refractory non\u2010degenerative monogenic dystonia is heterogeneous and long\u2010term results are sparse.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>The objective is to describe long\u2010term outcomes in a single\u2010center cohort and compare our results with a meta\u2010analysis cohort form literature.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>We performed a retrospective single\u2010center cohort study including consecutive pediatric patients with non\u2010degenerative genetic or idiopathic dystonia treated with globus pallidus internus deep brain stimulation at our center and a systematic review and individual\u2010patient data meta\u2010analysis with the same inclusion criteria. The primary outcome was the change from baseline in the Burke\u2010Fahn\u2010Marsden Dystonia Rating Scale\u2010movement (BFMDRS\u2010M) score.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>The clinical cohort included 25 patients with a mean study follow\u2010up of 11.4\u2009years. The meta\u2010analysis cohort included 224 patients with a mean follow\u2010up of 3\u2009years. Overall, the BFMDRS\u2010M mean improvements at 1\u2009year and at last follow\u2010up were 41% and 33% in the clinical cohort and 58.9% and 57.2% in the meta\u2010analysis cohort, respectively. TOR1A\u2010dystonia showed the greatest and most stable BFMDRS\u2010M improvement in both cohorts at 1\u2009year and at last follow\u2010up (76.3% and 74.3% in the clinical cohort; 69.6% and 67.3% in the meta\u2010analysis cohort), followed by SGCE\u2010dystonia (63% and 63.9% in the meta\u2010analysis cohort). THAP1\u2010dystonia (70.1% and 29.8% in the clinical cohort; 52.3% and 42.0% in the meta\u2010analysis cohort) and KMT2B\u2010dystonia (33.3% and 41.3% in the clinical cohort; 38.0% and 26.7% in the meta\u2010analysis cohort) showed a less pronounced or sustained response.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>\n                      Globus pallidus deep brain stimulation long\u2010term treatment seems effective with a possible gene\u2010specific differential effect. \u00a9 2024 The Authors.\n                      <jats:italic>Movement Disorders<\/jats:italic>\n                      published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/mds.29815","type":"journal-article","created":{"date-parts":[[2024,4,22]],"date-time":"2024-04-22T04:23:45Z","timestamp":1713759825000},"page":"1131-1144","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":23,"title":["Long\u2010Term\n                    <scp>Globus Pallidus Internus Deep Brain Stimulation<\/scp>\n                    in Pediatric Non\u2010Degenerative Dystonia: A Cohort Study and a Meta\u2010Analysis"],"prefix":"10.1002","volume":"39","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-6711-5406","authenticated-orcid":false,"given":"Valentina","family":"Duga","sequence":"first","affiliation":[{"name":"Child Neuropsychiatry Unit, Department of Pediatric Neuroscience Fondazione IRCCS Istituto Neurologico Carlo Besta  Milano Italy"},{"name":"Child and Adolescent Neuropsychiatric Unit, Foundation IRCCS Ca' Granda Ospedale Maggiore Policlinico  Milano 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