{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,27]],"date-time":"2026-07-27T13:01:40Z","timestamp":1785157300507,"version":"3.55.0"},"reference-count":31,"publisher":"Wiley","license":[{"start":{"date-parts":[[2026,7,27]],"date-time":"2026-07-27T00:00:00Z","timestamp":1785110400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,7,27]],"date-time":"2026-07-27T00:00:00Z","timestamp":1785110400000},"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":["Annals of Neurology"],"abstract":"<jats:sec>\n                    <jats:title>Objective<\/jats:title>\n                    <jats:p>\n                      <jats:italic>GNAO1<\/jats:italic>\n                      \u2010related disorder is a severe childhood\u2010onset hyperkinetic movement disorder punctuated by life\u2010threatening dyskinetic crises. Small series suggest benefit from bilateral globus pallidus internus deep brain stimulation (GPi\u2010DBS), but optimal timing, patient selection, long\u2010term outcomes, and genotype\u2010specific response remain unclear. We define the clinical impact of GPi\u2010DBS in the largest cohort assembled to date.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>\n                      Retrospective multicenter cohort study conducted through the DBSMatchMaker platform, including children and young adults with genetically confirmed\n                      <jats:italic>GNAO1<\/jats:italic>\n                      \u2010related disorder treated with bilateral GPi\u2010DBS across 17 centers. The primary outcome was change in dyskinetic crisis burden; secondary outcomes included BFMDRS, Clinical Global Impression (CGI), functional classifications, medication burden, and complications.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>\n                      Forty\u2010six patients underwent implantation at a mean age of 10.4\u2009\u00b1\u20094.8\u2009years with mean follow\u2010up of 4.5\u2009\u00b1\u20093.7\u2009years (longest\u2009=\u200917\u2009years). Over half (52.2%) were implanted emergently during dyskinetic status. Dyskinetic crises were reduced in 38 of 40 patients (95%) and intensive care unit (ICU) admissions in 86.1%. The Burke Fahn Marsden Dystonia Rating Scale (BFMDRS) motor scores improved by 27.5% (\n                      <jats:italic>p<\/jats:italic>\n                      \u2009&lt;\u20090.001, Cohen's d\u2009=\u20091.12); CGI was improved in 93.5%, with greater benefit for chorea than dystonia. Pain, sleep, and medication burden improved; functional classification was largely unchanged. Complications occurred in 19.6%, predominantly in emergent cases.\n                    <\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Interpretation<\/jats:title>\n                    <jats:p>\n                      Bilateral GPi\u2010DBS was associated with sustained prevention of dyskinetic crises and reduction of overall disease burden in\n                      <jats:italic>GNAO1<\/jats:italic>\n                      \u2010related disorder, with limited impact on functional classification. These findings support earlier elective neuromodulation before recurrent crises drive cumulative morbidity and identify GPi\u2010DBS as a crisis\u2010preventive, symptom\u2010modifying intervention rather than a restorative one. ANN NEUROL 2026\n                    <\/jats:p>\n                  <\/jats:sec>","DOI":"10.1002\/ana.78310","type":"journal-article","created":{"date-parts":[[2026,7,27]],"date-time":"2026-07-27T12:16:53Z","timestamp":1785154613000},"update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Outcomes of Bilateral Globus Pallidus Internus Deep Brain Stimulation in\n                    <scp>\n                      <i>GNAO1<\/i>\n                    <\/scp>\n                    \u2010Related Disorder: An International Multicenter Experience"],"prefix":"10.1002","author":[{"ORCID":"https:\/\/orcid.org\/0009-0004-3387-7623","authenticated-orcid":false,"given":"Katerina","family":"Bernardi","sequence":"first","affiliation":[{"name":"Movement Disorders Program, Department of Neurology and F.M. Kirby Neurobiology Center Boston Children's Hospital, Harvard Medical School  Boston MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0001-5277-5794","authenticated-orcid":false,"given":"Josh","family":"Rong","sequence":"additional","affiliation":[{"name":"Movement Disorders Program, Department of Neurology and F.M. Kirby Neurobiology Center Boston Children's Hospital, Harvard Medical School  Boston MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3019-1296","authenticated-orcid":false,"given":"Weston T.","family":"Northam","sequence":"additional","affiliation":[{"name":"Department of Neurosurgery Boston Children's Hospital, Harvard Medical School  Boston MA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Margaret","family":"Kami\u0144ska","sequence":"additional","affiliation":[{"name":"Complex Motor Disorder Service, Evelina London Children's Hospital, Guy's and St. Thomas\u2019 NHS Foundation Trust  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Harutomo","family":"Hasegawa","sequence":"additional","affiliation":[{"name":"Complex Motor Disorder Service, Evelina London Children's Hospital, Guy's and St. Thomas\u2019 NHS Foundation Trust  London UK"},{"name":"Department of Neurosurgery King's College Hospital NHS Foundation Trust  London UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7197-7391","authenticated-orcid":false,"given":"Jana","family":"Dom\u00ednguez\u2010Carral","sequence":"additional","affiliation":[{"name":"Movement Disorders Unit, Pediatric Neurology Department Institut de Recerca, Hospital Sant Joan de D\u00e9u Barcelona  Barcelona Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4878-8517","authenticated-orcid":false,"given":"Lindsey M.","family":"Vogt","sequence":"additional","affiliation":[{"name":"Division of Neurology and Division of Neurosurgery The Hospital for Sick Children, University of Toronto  Toronto Ontario Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joke M.","family":"Dijk","sequence":"additional","affiliation":[{"name":"Department of Pediatric Neurology Emma Children's Hospital, Amsterdam University Medical Center, Vrije Universiteit  Amsterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Martijn","family":"Beudel","sequence":"additional","affiliation":[{"name":"Department of Pediatric Neurology Emma Children's Hospital, Amsterdam University Medical Center, Vrije Universiteit  Amsterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eline","family":"Chauvet\u2010Piat","sequence":"additional","affiliation":[{"name":"Department of Pediatric Neurology Montpellier University Hospital  Montpellier France"},{"name":"Department of the Woman The Child, and the Adolescent, Division of Neurology, Geneva University Hospitals  Geneva Switzerland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emilie Chan","family":"Seng","sequence":"additional","affiliation":[{"name":"Department of Stereotactic and Functional Neurosurgery University Medical Center Gui de Chauliac, Montpellier University Hospital  Montpellier 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F.","family":"Jones","sequence":"additional","affiliation":[{"name":"Department of Pediatric Neurology Starship Children's Hospital  Auckland New Zealand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Beatriz","family":"De la Casa\u2010Fages","sequence":"additional","affiliation":[{"name":"Pediatric Neurology Unit, Department of Pediatrics Hospital General Universitario Gregorio Mara\u00f1\u00f3n  Madrid Spain"},{"name":"European Reference Network\u2010Rare Neurological Diseases  Brussels Belgium"},{"name":"Neuroscience Area, Instituto de Investigaci\u00f3n Sanitaria Gregorio Mara\u00f1\u00f3n  Madrid Spain"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mar\u00eda Concepci\u00f3n","family":"Miranda\u2010Herrero","sequence":"additional","affiliation":[{"name":"Pediatric Neurology Unit, Department of Pediatrics Hospital General Universitario Gregorio Mara\u00f1\u00f3n  Madrid Spain"},{"name":"European Reference Network\u2010Rare Neurological Diseases  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