{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,28]],"date-time":"2026-09-28T02:35:06Z","timestamp":1790562906050,"version":"4.1.0"},"reference-count":17,"publisher":"SAGE Publications","issue":"6","license":[{"start":{"date-parts":[[2018,4,16]],"date-time":"2018-04-16T00:00:00Z","timestamp":1523836800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["J Child Neurol"],"published-print":{"date-parts":[[2018,5]]},"abstract":"<jats:p>Dystonic storm or status dystonicus is a life-threatening hyperkinetic movement disorder with biochemical alterations due to the excessive muscle contractions. The medical management can require pediatric intensive care unit admission and a combination of medications while the underlying trigger is managed. Severe cases may require general anesthesia and paralytic agents with intubation and may relapse when these drugs are weaned. Deep brain stimulation of the globus pallidum has been reported to terminate dystonic storm in several pediatric cases. We present a 10-year-old boy with a de novo GNAO1 mutation\u2013induced dystonic storm who required a 2-month pediatric intensive care unit admission and remained refractory to all medical treatments. Deep brain stimulation was performed under general anesthetic without complication. His dyskinetic movements stopped with initiation of stimulation. He was discharged from the pediatric intensive care unit after 4 days. We present prospectively evaluated changes in dystonia symptoms and quality of life for a patient with GNAO1 mutation treated with deep brain stimulation.<\/jats:p>","DOI":"10.1177\/0883073818756134","type":"journal-article","created":{"date-parts":[[2018,4,16]],"date-time":"2018-04-16T21:07:11Z","timestamp":1523912831000},"page":"413-416","update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":45,"title":["<i>GNAO1<\/i>\n                    Mutation\u2013Induced Pediatric Dystonic Storm Rescue With Pallidal Deep Brain Stimulation"],"prefix":"10.1177","volume":"33","author":[{"given":"C. Michael","family":"Honey","sequence":"first","affiliation":[{"name":"Department of Surgery, Section of Neurosurgery, University of Manitoba, Winnipeg, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Armaan K.","family":"Malhotra","sequence":"additional","affiliation":[{"name":"Faculty of Medicine, University of British Columbia, Vancouver, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maja","family":"Tarailo-Graovac","sequence":"additional","affiliation":[{"name":"Centre for Molecular Medicine and Therapeutics, Vancouver, Canada"},{"name":"BC Children\u2019s Hospital Research Institute, University of British Columbia, Vancouver, Canada"},{"name":"Department of Medical Genetics, University of British Columbia, Vancouver, Canada"},{"name":"Institute of Physiology and Biochemistry, Faculty of Biology, University of Belgrade, Belgrade, Serbia"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Clara D. M.","family":"van Karnebeek","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, BC Children\u2019s Hospital Research Institute, Centre for Molecular Medicine and Therapeutics, University of British Columbia, Vancouver, Canada"},{"name":"Department of Pediatrics, Emma Children\u2019s Hospital, Academic Medical Centre, Amsterdam, the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gabriella","family":"Horvath","sequence":"additional","affiliation":[{"name":"BC Children\u2019s Hospital Research Institute, University of British Columbia, Vancouver, Canada"},{"name":"Division of Biochemical Diseases, Department of Pediatrics, University of British Columbia, Vancouver, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Adi","family":"Sulistyanto","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery, Department of Surgery, University of British Columbia, Vancouver, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2018,4,16]]},"reference":[{"key":"bibr1-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1016\/j.pediatrneurol.2016.02.018"},{"key":"bibr2-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1212\/NXG.0000000000000143"},{"key":"bibr3-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1177\/0883073815587945"},{"key":"bibr4-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1177\/0883073816666474"},{"key":"bibr5-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp-2017-315653"},{"key":"bibr6-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1111\/dmcn.12339"},{"key":"bibr7-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1038\/nature19057"},{"key":"bibr8-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1038\/ng.2892"},{"key":"bibr9-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1038\/nprot.2009.86"},{"key":"bibr10-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1002\/0471142905.hg0720s76"},{"key":"bibr11-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000004262"},{"key":"bibr12-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1159\/000446191"},{"key":"bibr13-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1007\/s00701-015-2514-5"},{"key":"bibr14-0883073818756134","first-page":"125","volume":"23","author":"Aydin S","year":"2013","journal-title":"Turk Neurosurg"},{"key":"bibr15-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1016\/S0021-9258(19)85162-1"},{"key":"bibr16-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1684\/epd.2017.0888"},{"key":"bibr17-0883073818756134","doi-asserted-by":"publisher","DOI":"10.1016\/j.cca.2015.10.011"}],"container-title":["Journal of Child Neurology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/0883073818756134","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/full-xml\/10.1177\/0883073818756134","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/journals.sagepub.com\/doi\/pdf\/10.1177\/0883073818756134","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,4,29]],"date-time":"2026-04-29T20:38:12Z","timestamp":1777495092000},"score":1,"resource":{"primary":{"URL":"https:\/\/journals.sagepub.com\/doi\/10.1177\/0883073818756134"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,4,16]]},"references-count":17,"journal-issue":{"issue":"6","published-print":{"date-parts":[[2018,5]]}},"alternative-id":["10.1177\/0883073818756134"],"URL":"https:\/\/doi.org\/10.1177\/0883073818756134","relation":{},"ISSN":["0883-0738","1708-8283"],"issn-type":[{"value":"0883-0738","type":"print"},{"value":"1708-8283","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,4,16]]}}}