{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,9]],"date-time":"2026-10-09T22:18:41Z","timestamp":1791584321119,"version":"4.3.4"},"reference-count":31,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,8,27]],"date-time":"2026-08-27T00:00:00Z","timestamp":1787788800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"},{"start":{"date-parts":[[2026,8,27]],"date-time":"2026-08-27T00:00:00Z","timestamp":1787788800000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Develop Med Child Neuro"],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Aim<\/jats:title>\n                    <jats:p>To examine longitudinal changes in self\u2010selected goals and dystonia severity over 1 year, 2 years, and 5\u2009years after deep brain stimulation (DBS) in children and young people (CYP) with dystonia and other hyperkinetic movement disorders, using CYP and caregiver reports.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Method<\/jats:title>\n                    <jats:p>The retrospective cohort study included follow\u2010up to 5\u2009years after DBS, with the number of participants varying across time points. In the CYP group, 62 participants were included at the 1\u2010year follow\u2010up, 50 at 2\u2009years, and 17 at 5\u2009years. In the parental and caregiver sample, 146 participants were included at 1\u2009year, 125 at 2\u2009years, and 51 at 5\u2009years. Changes across time were assessed using Wilcoxon signed\u2010rank tests, the Canadian Occupational Performance Measure (COPM), and Burke\u2013Fahn\u2013Marsden Dystonia Rating Scale \u2010 motor severity (BFMDRS\u2010m). Analyses were stratified by key characteristics of participants, including sex, age at surgery, Manual Ability Classification System (MACS) level, Gross Motor Function Classification System (GMFCS) level, and aetiology.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Median COPM performance increased from 2.8 to 6.1 (baseline to 5\u2009years), while COPM satisfaction improved from 2.6 to 7.6; parent\/caregiver reports showed similar trends. BFMDRS\u2010m scores decreased from 81.0 to 70.0, with the largest improvements in the first year. COPM changes were greater in monogenic dystonia and participants classified in MACS levels I to III. BFMDRS\u2010m improvements were more pronounced in monogenic dystonia, participants classified in GMFCS levels I to III, and those aged at least 12\u2009years.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Interpretation<\/jats:title>\n                    <jats:p>DBS showed sustained improvements in motor function and self\u2010selected goals, with consistent findings across CYP and caregiver reports. Functional gains were observed despite limited dystonia reduction, particularly in dystonic cerebral palsy.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/dmcn.70481","type":"journal-article","created":{"date-parts":[[2026,8,27]],"date-time":"2026-08-27T15:43:17Z","timestamp":1787845397000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Deep brain stimulation outcomes and management of childhood\u2010onset dystonia and associated hyperkinetic movements disorders"],"prefix":"10.1111","author":[{"ORCID":"https:\/\/orcid.org\/0009-0008-3946-3375","authenticated-orcid":false,"given":"Sinead","family":"Barkey","sequence":"first","affiliation":[{"name":"Complex Motor Disorders Service, Evelina London Children's Hospital Guy's &amp; St Thomas' NHS Foundation Trust  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laura","family":"Ainaga","sequence":"additional","affiliation":[{"name":"Complex Motor Disorders Service, Evelina London Children's Hospital Guy's &amp; St Thomas' NHS Foundation Trust  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1494-5676","authenticated-orcid":false,"given":"Eva Maria","family":"Navarrete Mu\u00f1oz","sequence":"additional","affiliation":[{"name":"Occupational Therapy Research Group (InTeO, Investigaci\u00f3n en Terapia Ocupacional) Miguel Hern\u00e1ndez University  Alicante Spain"},{"name":"Institute for Health and Biomedical Research of Alicante (Instituto de Investigaci\u00f3n Sanitaria y Biom\u00e9dica de Alicante, ISABIAL)  Alicante Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5180-4242","authenticated-orcid":false,"given":"Verity","family":"McClelland","sequence":"additional","affiliation":[{"name":"Complex Motor Disorders Service, Evelina London Children's Hospital Guy's &amp; St Thomas' NHS Foundation Trust  London UK"},{"name":"Department of Basic and Clinical Neuroscience Institute of Psychiatry, Psychology and Neurosciences, King's College London  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Keyoumars","family":"Ashkan","sequence":"additional","affiliation":[{"name":"Functional Neurosurgery, King's College Hospital NHS Foundation Trust  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Richard","family":"Selway","sequence":"additional","affiliation":[{"name":"Functional Neurosurgery, King's College Hospital NHS Foundation Trust  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3711-6790","authenticated-orcid":false,"given":"Harutomo","family":"Hasegawa","sequence":"additional","affiliation":[{"name":"Complex Motor Disorders Service, Evelina London Children's Hospital Guy's &amp; St Thomas' NHS Foundation Trust  London UK"},{"name":"Functional Neurosurgery, King's College Hospital NHS Foundation Trust  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5524-6177","authenticated-orcid":false,"given":"Daniel E","family":"Lumsden","sequence":"additional","affiliation":[{"name":"Complex Motor Disorders Service, Evelina London Children's Hospital Guy's &amp; St Thomas' NHS Foundation Trust  London UK"},{"name":"Research Department of Early Life Imaging Biomedical Engineering and Imaging Sciences, King's College London  UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9314-2259","authenticated-orcid":false,"given":"Jean\u2010Pierre","family":"Lin","sequence":"additional","affiliation":[{"name":"Complex Motor Disorders Service, Evelina London Children's Hospital Guy's &amp; St Thomas' NHS Foundation Trust  London UK"},{"name":"Department of Women and Children's Health School of Life Course &amp; Population Sciences, King's College London  UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-1488-6415","authenticated-orcid":false,"given":"Hortensia","family":"Gimeno","sequence":"additional","affiliation":[{"name":"Complex Motor Disorders Service, Evelina London Children's Hospital Guy's &amp; St Thomas' NHS Foundation Trust  London UK"},{"name":"Childhood Participation Lab, Centre for Bone and Joint Health Blizard Institute, Queen Mary University of London  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,8,27]]},"reference":[{"key":"e_1_2_11_2_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.30220"},{"key":"e_1_2_11_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.23088"},{"key":"e_1_2_11_4_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp-2013-307041"},{"key":"e_1_2_11_5_1","doi-asserted-by":"crossref","DOI":"10.1111\/dmcn.14874","article-title":"Pharmacological and neurosurgical interventions for individuals with cerebral palsy and dystonia: a systematic review update and meta\u2010analysis","author":"Bohn E","year":"2021","journal-title":"Dev Med Child Neurol."},{"key":"e_1_2_11_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.15921"},{"key":"e_1_2_11_7_1","first-page":"1","article-title":"Intervention outcomes of children with non\u2010degenerative dystonia and associated hyperkinetic movement disorders: A scoping review","volume":"00","author":"Gimeno H","year":"2026","journal-title":"Developmental Medicine & Child Neurology."},{"key":"e_1_2_11_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/ene.13255"},{"key":"e_1_2_11_9_1","doi-asserted-by":"publisher","DOI":"10.1007\/s10143-018-1047-9"},{"key":"e_1_2_11_10_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jns.2024.122970"},{"key":"e_1_2_11_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.14063"},{"key":"e_1_2_11_12_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.35.1.73"},{"key":"e_1_2_11_13_1","doi-asserted-by":"publisher","DOI":"10.1159\/000524577"},{"key":"e_1_2_11_14_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.29815"},{"key":"e_1_2_11_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2011.12.014"},{"key":"e_1_2_11_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2016.08.016"},{"key":"e_1_2_11_17_1","doi-asserted-by":"publisher","DOI":"10.5334\/tohm.69"},{"key":"e_1_2_11_18_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2012.07.007"},{"key":"e_1_2_11_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2015.01.003"},{"key":"e_1_2_11_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2013.12.010"},{"key":"e_1_2_11_21_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.28898"},{"key":"e_1_2_11_22_1","volume-title":"Canadian Occupational Performance Measure","author":"Law M","year":"2014"},{"key":"e_1_2_11_23_1","doi-asserted-by":"crossref","DOI":"10.1016\/j.ejpn.2021.04.009","article-title":"Rehabilitation in childhood\u2010onset hyperkinetic movement disorders including dystonia: Treatment change in outcomes across the ICF and feasibility of outcomes for full trial evaluation","author":"Gimeno H","year":"2021","journal-title":"Eur J Paediatr Neurol."},{"key":"e_1_2_11_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.parkreldis.2016.10.001"},{"key":"e_1_2_11_25_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.parkreldis.2022.11.006"},{"key":"e_1_2_11_26_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp-2015-311803"},{"key":"e_1_2_11_27_1","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.12117"},{"key":"e_1_2_11_28_1","doi-asserted-by":"crossref","DOI":"10.1016\/j.ejpn.2012.12.007","article-title":"Improvement in upper limb function in children with dystonia following deep brain stimulation","author":"Gimeno H","year":"2013","journal-title":"Eur J Paediatr Neurol."},{"key":"e_1_2_11_29_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2019.02.005"},{"key":"e_1_2_11_30_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000007092"},{"key":"e_1_2_11_31_1","first-page":"8","article-title":"Cognitive strategy training in childhood\u2010onset movement disorders. 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