{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,9]],"date-time":"2026-10-09T22:18:46Z","timestamp":1791584326145,"version":"4.3.4"},"reference-count":44,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2018,10,15]],"date-time":"2018-10-15T00:00:00Z","timestamp":1539561600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Develop Med Child Neuro"],"published-print":{"date-parts":[[2019,1]]},"abstract":"<jats:sec><jats:title>Aim<\/jats:title><jats:p>We performed a meta\u2010analysis with individual participant data of deep brain stimulation (<jats:styled-content style=\"fixed-case\">DBS<\/jats:styled-content>) for dystonia in children and young people.<\/jats:p><\/jats:sec><jats:sec><jats:title>Method<\/jats:title><jats:p>Three databases (PubMed, Embase, and Web of Science) were queried from January 1999 to August 2017 with no language restrictions to identify case studies and cohort studies reporting on pediatric patients (age \u226421y) with dystonia. The primary outcomes were changes in Burke\u2010Fahn\u2010Marsden (<jats:styled-content style=\"fixed-case\">BFM<\/jats:styled-content>) or Barry\u2010Albright Dystonia Scale scores. A mixed\u2010effects regression was used to identify associations between clinical covariates and outcomes.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Of 2509 citations reviewed, 72 articles (321 children) were eligible. At last follow\u2010up (median 12mo, 25th centile=9.0; 75th centile=32.2), 277 (86.3%) patients showed improvement in dystonia, while 66.1 percent showed clinically significant (&gt;20%) <jats:styled-content style=\"fixed-case\">BFM<\/jats:styled-content> Dystonia Rating Scale\u2010motor improvement. On multivariable hierarchical regression, older age at dystonia onset, inherited dystonia without nervous system pathology and idiopathic dystonia (vs inherited with nervous system pathology or acquired dystonia), and truncal involvement indicated a better outcome (<jats:italic>p<\/jats:italic>&lt;0.05).<\/jats:p><\/jats:sec><jats:sec><jats:title>Interpretation<\/jats:title><jats:p>The data suggest that <jats:styled-content style=\"fixed-case\">DBS<\/jats:styled-content> is effective and should be considered in selected children with inherited or idiopathic dystonia.<\/jats:p><\/jats:sec><jats:sec><jats:title>What this paper adds<\/jats:title><jats:p>\n<jats:list list-type=\"bullet\">\n\n<jats:list-item><jats:p>Deep brain stimulation is effective in selected children with inherited or idiopathic dystonia.<\/jats:p><\/jats:list-item>\n<\/jats:list>\n<\/jats:p><\/jats:sec>","DOI":"10.1111\/dmcn.14063","type":"journal-article","created":{"date-parts":[[2018,10,15]],"date-time":"2018-10-15T10:39:32Z","timestamp":1539599972000},"page":"49-56","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":120,"title":["Deep brain stimulation for pediatric dystonia: a meta\u2010analysis with individual participant data"],"prefix":"10.1111","volume":"61","author":[{"given":"Lior M","family":"Elkaim","sequence":"first","affiliation":[{"name":"Faculty of Medicine Universit\u00e9 de Montr\u00e9al Montreal Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Naif M","family":"Alotaibi","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery Department of Surgery University of Toronto Toronto Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alissa","family":"Sigal","sequence":"additional","affiliation":[{"name":"Faculty of Medicine Universit\u00e9 de Montr\u00e9al Montreal Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Haifa M","family":"Alotaibi","sequence":"additional","affiliation":[{"name":"College of Medicine King Saud University Riyadh Saudi Arabia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nir","family":"Lipsman","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery Department of Surgery University of Toronto Toronto Canada"},{"name":"Division of Neurosurgery Sunnybrook Health Sciences Centre Toronto Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Suneil K","family":"Kalia","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery Department of Surgery University of Toronto Toronto Canada"},{"name":"Division of Neurosurgery Krembil Neuroscience Centre Toronto Western Hospital Toronto Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Darcy L","family":"Fehlings","sequence":"additional","affiliation":[{"name":"Child Development Program Holland Bloorview Rehabilitation Hospital University of Toronto Toronto Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Andres M","family":"Lozano","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery Department of Surgery University of Toronto Toronto Canada"},{"name":"Division of Neurosurgery Krembil Neuroscience Centre Toronto Western Hospital Toronto Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9068-8184","authenticated-orcid":false,"given":"George M","family":"Ibrahim","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery Department of Surgery University of Toronto Toronto Canada"},{"name":"Division of Neurosurgery The Hospital for Sick Children Program in Neuroscience and Mental Health The Hospital for Sick Children Research Institute Toronto Canada"},{"name":"Department of Surgery Institute of Biomaterials and Biomedical Engineering Institute of Medical Science University of Toronto Toronto Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"North American Pediatric DBS Collaboration","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2018,10,15]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2016.08.010"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2016.02.003"},{"key":"e_1_2_8_4_1","first-page":"139","article-title":"Traitement de la dystonie g\u00e9n\u00e9ralis\u00e9e \u00e0 d\u00e9but pr\u00e9coce par stimulation chronique bilat\u00e9rale des globus pallidus internes. A propos d'un cas","volume":"45","author":"Coubes P","year":"1999","journal-title":"Neurochirurgie"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.3171\/2011.8.PEDS11153"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp-2011-301714"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.braindev.2009.03.010"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1227\/NEU.0b013e3182077396"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp-2013-307041"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.3171\/2014.8.PEDS141"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pmed.1000097"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.35.1.73"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1469-8749.1999.tb00626.x"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.25475"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1016\/0197-2456(86)90046-2"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.22515"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa042187"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1136\/adc.2006.095380"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp.2010.207993"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jocn.2015.08.034"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2011.12.014"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1227\/01.NEU.0000108643.94730.21"},{"key":"e_1_2_8_23_1","doi-asserted-by":"publisher","DOI":"10.3171\/2013.8.FOCUS13300"},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.23349"},{"key":"e_1_2_8_25_1","doi-asserted-by":"publisher","DOI":"10.1002\/ana.20457"},{"key":"e_1_2_8_26_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.25339"},{"key":"e_1_2_8_27_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1474-4422(17)30252-1"},{"key":"e_1_2_8_28_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2012.07.007"},{"key":"e_1_2_8_29_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2013.12.010"},{"key":"e_1_2_8_30_1","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.12339"},{"key":"e_1_2_8_31_1","doi-asserted-by":"publisher","DOI":"10.3171\/2011.10.PEDS11360"},{"key":"e_1_2_8_32_1","doi-asserted-by":"publisher","DOI":"10.1159\/000446191"},{"key":"e_1_2_8_33_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2016.07.024"},{"key":"e_1_2_8_34_1","doi-asserted-by":"publisher","DOI":"10.1002\/mds.22433"},{"key":"e_1_2_8_35_1","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.12117"},{"key":"e_1_2_8_36_1","doi-asserted-by":"publisher","DOI":"10.1093\/brain\/awr122"},{"key":"e_1_2_8_37_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.parkreldis.2016.07.018"},{"key":"e_1_2_8_38_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1525-1403.2006.00067.x"},{"key":"e_1_2_8_39_1","doi-asserted-by":"publisher","DOI":"10.1111\/ene.13255"},{"key":"e_1_2_8_40_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2016.06.002"},{"key":"e_1_2_8_41_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2016.05.023"},{"key":"e_1_2_8_42_1","doi-asserted-by":"publisher","DOI":"10.4172\/2167-0870.1000317"},{"key":"e_1_2_8_43_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199708213370810"},{"key":"e_1_2_8_44_1","doi-asserted-by":"publisher","DOI":"10.1093\/oxfordjournals.aje.a117326"},{"key":"e_1_2_8_45_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ejpn.2016.08.016"}],"container-title":["Developmental Medicine &amp; Child Neurology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fdmcn.14063","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/dmcn.14063","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/dmcn.14063","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/dmcn.14063","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,9,10]],"date-time":"2023-09-10T17:25:46Z","timestamp":1694366746000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/dmcn.14063"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,10,15]]},"references-count":44,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2019,1]]}},"alternative-id":["10.1111\/dmcn.14063"],"URL":"https:\/\/doi.org\/10.1111\/dmcn.14063","archive":["Portico"],"relation":{},"ISSN":["0012-1622","1469-8749"],"issn-type":[{"value":"0012-1622","type":"print"},{"value":"1469-8749","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,10,15]]},"assertion":[{"value":"2018-08-28","order":1,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2018-10-15","order":2,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}