{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,30]],"date-time":"2026-05-30T09:06:22Z","timestamp":1780131982406,"version":"3.54.0"},"reference-count":48,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"6","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,6]]},"abstract":"\n OBJECTIVE<\/jats:title>\n Dystonia is among the most common pediatric movement disorders and can manifest with a range of debilitating symptoms, including sleep disruptions. The duration and quality of sleep are strongly associated with quality of life in these individuals and could serve as biomarkers of dystonia severity and the efficacy of interventions such as deep brain stimulation (DBS). Thus, this study investigated sleep duration and its relationship to disease severity and DBS response in pediatric dystonia.<\/jats:p>\n <\/jats:sec>\n \n METHODS<\/jats:title>\n Actigraphs (wearable three-axis accelerometers) were used to record multiday sleep data in 22 children with dystonia, including 6 patients before and after DBS implantation, and age- and sex- matched healthy controls. Data were preprocessed, and metrics of sleep duration and quality were extracted. Repeated-measures statistical analyses were used.<\/jats:p>\n <\/jats:sec>\n \n RESULTS<\/jats:title>\n Children with dystonia slept less than typically developing children (p = 0.009), and shorter sleep duration showed trending correlation with worse dystonia severity (r = \u22120.421, p = 0.073). Of 4 patients who underwent DBS and had good-quality data, 1 demonstrated significantly improved sleep (p < 0.001) postoperatively. Reduction in dystonia severity strongly correlated with increased sleep duration after DBS implantation (r = \u22120.965, p = 0.035).<\/jats:p>\n <\/jats:sec>\n \n CONCLUSIONS<\/jats:title>\n Sleep disturbances are an underrecognized marker of pediatric dystonia severity, as well as the effectiveness of interventions such as DBS. They can serve as objective biomarkers of disease burden and symptom progression after treatment.<\/jats:p>\n <\/jats:sec>","DOI":"10.3171\/2024.3.focus2462","type":"journal-article","created":{"date-parts":[[2024,6,1]],"date-time":"2024-06-01T05:41:52Z","timestamp":1717220512000},"page":"E17","source":"Crossref","is-referenced-by-count":0,"title":["Actigraph-based quantification of sleep in children with dystonia undergoing deep brain stimulation"],"prefix":"10.3171","volume":"56","author":[{"given":"Frederick","family":"Zhang","sequence":"first","affiliation":[{"name":"Faculty of Medicine, University of Ottawa, Ontario;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karim","family":"Mithani","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery, The Hospital for Sick Children, Toronto, Ontario;"},{"name":"Institute of Biomedical Engineering, University of Toronto, Ontario; and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sara","family":"Breitbart","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery, The Hospital for Sick Children, Toronto, Ontario;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Han","family":"Yan","sequence":"additional","affiliation":[{"name":"Division of Neurosurgery, The Hospital for Sick Children, Toronto, Ontario;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alfonso","family":"Fasano","sequence":"additional","affiliation":[{"name":"Edmond J. Safra Program in Parkinson\u2019s Disease, Morton and Gloria Shulman Movement Disorders Clinic, Toronto Western Hospital, Toronto, Ontario;"},{"name":"Division of Neurology, University of Toronto, Ontario;"},{"name":"Krembil Brain Institute, University Health Network, Toronto, Ontario;"},{"name":"Center for Advancing Neurotechnological Innovation to Application (CRANIA), University Health Network, Toronto, Ontario;"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"George M.","family":"Ibrahim","sequence":"additional","affiliation":[{"name":"Departments of Surgery and"},{"name":"Institute of Biomedical Engineering, University of Toronto, Ontario; and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Carolina","family":"Gorodetsky","sequence":"additional","affiliation":[{"name":"Division of Neurology, The Hospital for Sick Children, Toronto, Ontario, Canada"},{"name":"Pediatrics, University of Toronto, Ontario;"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1754","reference":[{"issue":"7","key":"b1","series-title":"Mov Disord","first-page":"921","article-title":"Special concerns in defining, studying, and treating dystonia in children","volume":"28","author":"Mink JW","year":"2013","unstructured":"Mink JW. 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Mov Disord. 2013;28(7):921\u2013925."},{"issue":"7","key":"b2","series-title":"Mov Disord","first-page":"1206","article-title":"Nonmotor manifestations of dystonia: a systematic review","volume":"26","author":"Kuyper DJ","year":"2011","unstructured":"Kuyper DJ, Parra V, Aerts S, Okun MS, Kluger BM. Nonmotor manifestations of dystonia: a systematic review. Mov Disord. 2011;26(7):1206\u20131217."},{"key":"b3","series-title":"Sleep Med Rev","first-page":"95","article-title":"Sleep in patients with primary dystonia: a systematic review on the state of research and perspectives","volume":"26","author":"Hertenstein E","year":"2016","unstructured":"Hertenstein E, Tang NK, Bernstein CJ, Nissen C, Underwood MR, Sandhu HK. Sleep in patients with primary dystonia: a systematic review on the state of research and perspectives. 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Dev Med Child Neurol. 2019;61(1):49\u201356."},{"issue":"9","key":"b6","series-title":"Lancet Neurol","first-page":"741","article-title":"Clinical presentation and management of dyskinetic cerebral palsy","volume":"16","author":"Monbaliu E","year":"2017","unstructured":"Monbaliu E, Himmelmann K, Lin JP. Clinical presentation and management of dyskinetic cerebral palsy. Lancet Neurol. 2017;16(9):741\u2013749."},{"key":"b7","series-title":"Sleep Med Rev","first-page":"63","article-title":"Sleep disorders in children with cerebral palsy: an integrative review","volume":"30","author":"L\u00e9lis AL","year":"2016","unstructured":"L\u00e9lis AL, Cardoso MV, Hall WA. Sleep disorders in children with cerebral palsy: an integrative review. 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J Neurosurg Pediatr. 2019;24(4):442\u2013450."},{"issue":"3","key":"b12","series-title":"Neurosurg Rev","first-page":"873","article-title":"Deep brain stimulation in pediatric dystonia: a systematic review","volume":"43","author":"Hale AT","year":"2020","unstructured":"Hale AT, Monsour MA, Rolston JD, Naftel RP, Englot DJ. Deep brain stimulation in pediatric dystonia: a systematic review. Neurosurg Rev. 2020;43(3):873\u2013880."},{"issue":"1","key":"b13","series-title":"Neurology","first-page":"73","article-title":"Validity and reliability of a rating scale for the primary torsion dystonias","volume":"35","author":"Burke RE","year":"1985","unstructured":"Burke RE, Fahn S, Marsden CD, Bressman SB, Moskowitz C, Friedman J. Validity and reliability of a rating scale for the primary torsion dystonias. Neurology. 1985;35(1):73\u201377."},{"issue":"3","key":"b14","series-title":"J Meas Phys Behav","first-page":"188","article-title":"GGIR: A research community\u2013driven open source R package for generating physical activity and sleep outcomes from multi-day raw accelerometer data","volume":"2","author":"Migueles JH","year":"2019","unstructured":"Migueles JH, Rowlands AV, Huber F, Sabia S, van Hees VT. GGIR: A research community\u2013driven open source R package for generating physical activity and sleep outcomes from multi-day raw accelerometer data. J Meas Phys Behav. 2019;2(3):188\u2013196."},{"issue":"9","key":"b15","series-title":"Med Sci Sports Exerc","first-page":"1816","article-title":"Age group comparability of raw accelerometer output from wrist- and hip-worn monitors","volume":"46","author":"Hildebrand M","year":"2014","unstructured":"Hildebrand M, van Hees VT, Hansen BH, Ekelund U. Age group comparability of raw accelerometer output from wrist- and hip-worn monitors. Med Sci Sports Exerc. 2014;46(9):1816\u20131824."},{"issue":"11","key":"b16","series-title":"PLoS One","first-page":"e0142533","article-title":"A novel, open access method to assess sleep duration using a wrist-worn accelerometer","volume":"10","author":"van Hees VT","year":"2015","unstructured":"van Hees VT, Sabia S, Anderson KN, A novel, open access method to assess sleep duration using a wrist-worn accelerometer. PLoS One. 2015;10(11):e0142533."},{"issue":"1","key":"b17","series-title":"Sci Rep","first-page":"12975","article-title":"Estimating sleep parameters using an accelerometer without sleep diary","volume":"8","author":"van Hees VT","year":"2018","unstructured":"van Hees VT, Sabia S, Jones SE. Estimating sleep parameters using an accelerometer without sleep diary. Sci Rep. 2018;8(1):12975."},{"issue":"7","key":"b18","series-title":"Mov Disord","first-page":"1218","article-title":"Minimal clinically important differences for Burke-Fahn-Marsden Dystonia Rating Scale and 36-Item Short-Form Health Survey","volume":"35","author":"Pint\u00e9r D","year":"2020","unstructured":"Pint\u00e9r D, Janszky J, Kov\u00e1cs N. Minimal clinically important differences for Burke-Fahn-Marsden Dystonia Rating Scale and 36-Item Short-Form Health Survey. Mov Disord. 2020;35(7):1218\u20131223."},{"issue":"2","key":"b19","series-title":"Sleep Med","first-page":"213","article-title":"Sleep disorders in children with cerebral palsy: neurodevelopmental and behavioral correlates","volume":"15","author":"Romeo DM","year":"2014","unstructured":"Romeo DM, Brogna C, Quintiliani M, Sleep disorders in children with cerebral palsy: neurodevelopmental and behavioral correlates. 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