{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,1]],"date-time":"2026-06-01T07:03:35Z","timestamp":1780297415256,"version":"3.54.0"},"reference-count":36,"publisher":"Elsevier BV","issue":"3","license":[{"start":{"date-parts":[[2026,6,1]],"date-time":"2026-06-01T00:00:00Z","timestamp":1780272000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/tdm\/userlicense\/1.0\/"},{"start":{"date-parts":[[2026,6,1]],"date-time":"2026-06-01T00:00:00Z","timestamp":1780272000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/www.elsevier.com\/legal\/tdmrep-license"},{"start":{"date-parts":[[2026,5,7]],"date-time":"2026-05-07T00:00:00Z","timestamp":1778112000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":["brainanddevelopment.com","clinicalkey.com","clinicalkey.com.au","clinicalkey.es","clinicalkey.fr","clinicalkey.jp","elsevier.com","sciencedirect.com"],"crossmark-restriction":true},"short-container-title":["Brain and Development"],"published-print":{"date-parts":[[2026,6]]},"DOI":"10.1016\/j.braindev.2026.104545","type":"journal-article","created":{"date-parts":[[2026,5,12]],"date-time":"2026-05-12T16:04:32Z","timestamp":1778601872000},"page":"104545","update-policy":"https:\/\/doi.org\/10.1016\/elsevier_cm_policy","source":"Crossref","is-referenced-by-count":0,"title":["Genotype-dependent response to combined levodopa\/benserazide and trihexyphenidyl in pediatric genetic dystonia"],"prefix":"10.1016","volume":"48","author":[{"given":"Xiaolin","family":"Zhou","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yu","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiangyang","family":"Luo","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhanwen","family":"He","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mujin","family":"Liu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pinggan","family":"Li","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"78","reference":[{"issue":"10","key":"10.1016\/j.braindev.2026.104545_bb0005","doi-asserted-by":"crossref","first-page":"3755","DOI":"10.1007\/s10803-024-06467-3","article-title":"Dyskinetic CP in children: clinical perspectives on common comorbidities and health-related quality of life","volume":"55","author":"Saini","year":"2024","journal-title":"J Autism Dev Disord"},{"key":"10.1016\/j.braindev.2026.104545_bb0010","article-title":"Dystonia in cerebral palsy; what are the treatment options?","volume":"166","author":"van de Pol","year":"2022","journal-title":"Ned Tijdschr Geneeskd"},{"issue":"3","key":"10.1016\/j.braindev.2026.104545_bb0015","doi-asserted-by":"crossref","first-page":"262","DOI":"10.1016\/j.jmoldx.2021.12.003","article-title":"A clinical and integrated genetic study of isolated and combined dystonia in Taiwan","volume":"24","author":"Wu","year":"2022","journal-title":"J Mol Diagn"},{"issue":"3","key":"10.1016\/j.braindev.2026.104545_bb0020","doi-asserted-by":"crossref","first-page":"8","DOI":"10.1007\/s11910-021-01095-1","article-title":"Genetic dystonias: update on classification and new genetic discoveries","volume":"21","author":"Keller Sarmiento","year":"2021","journal-title":"Curr Neurol Neurosci Rep"},{"issue":"9","key":"10.1016\/j.braindev.2026.104545_bb0025","doi-asserted-by":"crossref","first-page":"2005","DOI":"10.1002\/mds.28642","article-title":"Rational design of novel therapies for pantothenate kinase-associated neurodegeneration","volume":"36","author":"Thakur","year":"2021","journal-title":"Mov Disord"},{"issue":"5","key":"10.1016\/j.braindev.2026.104545_bb0030","first-page":"384","article-title":"Progress in molecular genetics of epilepsy with myoclonic-atonic seizure","volume":"57","author":"Chen","year":"2019","journal-title":"Zhonghua Er Ke Za Zhi"},{"issue":"2","key":"10.1016\/j.braindev.2026.104545_bb0035","doi-asserted-by":"crossref","first-page":"257","DOI":"10.1016\/j.ejpn.2017.12.009","article-title":"ATP1A3-related disorders: an update","volume":"22","author":"Carecchio","year":"2018","journal-title":"Eur J Paediatr Neurol"},{"issue":"11","key":"10.1016\/j.braindev.2026.104545_bb0040","doi-asserted-by":"crossref","first-page":"3968","DOI":"10.1093\/brain\/awac001","article-title":"DYT6 mutated THAP1 is a cell type dependent regulator of the SP1 family","volume":"145","author":"Cheng","year":"2022","journal-title":"Brain"},{"issue":"4","key":"10.1016\/j.braindev.2026.104545_bb0045","doi-asserted-by":"crossref","first-page":"405","DOI":"10.1007\/s00702-020-02268-x","article-title":"Isolated dystonia: clinical and genetic updates","volume":"128","author":"Domingo","year":"2021","journal-title":"J Neural Transm (Vienna)"},{"issue":"4","key":"10.1016\/j.braindev.2026.104545_bb0050","doi-asserted-by":"crossref","first-page":"500","DOI":"10.1097\/WCO.0000000000000831","article-title":"Recent genetic advances in early-onset dystonia","volume":"33","author":"Steel","year":"2020","journal-title":"Curr Opin Neurol"},{"issue":"8","key":"10.1016\/j.braindev.2026.104545_bb0055","doi-asserted-by":"crossref","first-page":"1222","DOI":"10.1111\/jpc.15429","article-title":"National surveillance of oral medication prescription for children with dystonic cerebral palsy","volume":"57","author":"Harvey","year":"2021","journal-title":"J Paediatr Child Health"},{"issue":"1","key":"10.1016\/j.braindev.2026.104545_bb0060","first-page":"115","article-title":"Trihexyphenidyl in young children with dystonic cerebral palsy: a single arm study","volume":"16","author":"Rajkumar","year":"2023","journal-title":"J Pediatr Rehabil Med"},{"issue":"10","key":"10.1016\/j.braindev.2026.104545_bb0065","doi-asserted-by":"crossref","first-page":"1280","DOI":"10.1111\/dmcn.15572","article-title":"Overview of Cochrane systematic reviews for rehabilitation interventions in individuals with cerebral palsy: a mapping synthesis","volume":"65","author":"Liguori","year":"2023","journal-title":"Dev Med Child Neurol"},{"issue":"2","key":"10.1016\/j.braindev.2026.104545_bb0070","doi-asserted-by":"crossref","first-page":"81","DOI":"10.4103\/aomd.aomd_55_21","article-title":"A clinical approach to the patients with combination of dystonia and myoclonus","volume":"5","author":"Chouksey","year":"2022","journal-title":"Ann Mov Disord"},{"key":"10.1016\/j.braindev.2026.104545_bb0075","doi-asserted-by":"crossref","first-page":"36","DOI":"10.1016\/j.ejpn.2022.06.015","article-title":"Dystonia during hand activity in children with spastic unilateral cerebral palsy, an observational study","volume":"41","author":"Tedroff","year":"2022","journal-title":"Eur J Paediatr Neurol"},{"key":"10.1016\/j.braindev.2026.104545_bb0080","doi-asserted-by":"crossref","first-page":"1707423","DOI":"10.3389\/fneur.2026.1707423","article-title":"Efficacy of Madopar and trihexyphenidyl combination therapy for dystonia in children with cerebral palsy","volume":"17","author":"Zhou","year":"2026","journal-title":"Front Neurol"},{"issue":"8","key":"10.1016\/j.braindev.2026.104545_bb0085","doi-asserted-by":"crossref","first-page":"1136","DOI":"10.1002\/mdc3.13522","article-title":"Prolonged episodic dystonia in tyrosine hydroxylase deficiency due to homozygous c.698G>A (p.Arg233His) mutation\u2014a diagnostic challenge","volume":"9","author":"Panda","year":"2022","journal-title":"Mov Disord Clin Pract"},{"key":"10.1016\/j.braindev.2026.104545_bb0090","doi-asserted-by":"crossref","first-page":"48","DOI":"10.1016\/j.parkreldis.2021.08.019","article-title":"Dopaminergic and serotonergic alterations in plasma in three groups of dystonia patients","volume":"91","author":"Timmers","year":"2021","journal-title":"Parkinsonism Relat Disord"},{"issue":"12","key":"10.1016\/j.braindev.2026.104545_bb0095","doi-asserted-by":"crossref","first-page":"1555","DOI":"10.1080\/14656566.2021.1919083","article-title":"Perspectives on the pharmacological management of dystonia","volume":"22","author":"Svetel","year":"2021","journal-title":"Expert Opin Pharmacother"},{"issue":"1","key":"10.1016\/j.braindev.2026.104545_bb0100","doi-asserted-by":"crossref","first-page":"311","DOI":"10.1186\/s13023-022-02465-9","article-title":"Therapeutic approach with commercial supplements for pantothenate kinase-associated neurodegeneration with residual PANK2 expression levels","volume":"17","author":"\u00c1lvarez-C\u00f3rdoba","year":"2022","journal-title":"Orphanet J Rare Dis"},{"issue":"3","key":"10.1016\/j.braindev.2026.104545_bb0105","doi-asserted-by":"crossref","first-page":"26","DOI":"10.1007\/s11910-017-0735-0","article-title":"Update on the genetics of dystonia","volume":"17","author":"Lohmann","year":"2017","journal-title":"Curr Neurol Neurosci Rep"},{"key":"10.1016\/j.braindev.2026.104545_bb0110","doi-asserted-by":"crossref","first-page":"105","DOI":"10.1016\/j.ebiom.2024.105108","article-title":"Impacts of gene variants on drug effects\u2014the foundation of genotype-guided pharmacologic therapy for long QT syndrome and short QT syndrome","volume":"103","author":"Zhao","year":"2024","journal-title":"EBioMedicine"},{"issue":"2","key":"10.1016\/j.braindev.2026.104545_bb0115","doi-asserted-by":"crossref","first-page":"1","DOI":"10.59298\/ROJPHM\/2024\/3214","article-title":"The role of genomics in predicting drug response","volume":"3","author":"Nyiramana","year":"2024","journal-title":"ROJPHM"},{"issue":"8","key":"10.1016\/j.braindev.2026.104545_bb0120","doi-asserted-by":"crossref","first-page":"274","DOI":"10.1016\/j.braindev.2024.06.002","article-title":"Epidemiological study on pediatric-onset dystonia in Japan: a questionnaire-based survey","volume":"46","author":"Suenaga","year":"2024","journal-title":"Brain Dev"},{"issue":"60\u201361","key":"10.1016\/j.braindev.2026.104545_bb0125","doi-asserted-by":"crossref","first-page":"54","DOI":"10.21857\/mnlqgcr6dy","article-title":"Dystonia and deep brain stimulation: correlation between genetic mutation and clinical outcome","volume":"553","author":"Ro\u017emari\u0107","year":"2022","journal-title":"Rad Hrvatske Akademije Znanosti i Umjetnosti Medicinske Znanosti"},{"issue":"2","key":"10.1016\/j.braindev.2026.104545_bb0130","doi-asserted-by":"crossref","first-page":"207","DOI":"10.1111\/dmcn.15298","article-title":"Early recognition of SGCE-myoclonus-dystonia in children","volume":"65","author":"Correa-Vela","year":"2023","journal-title":"Dev Med Child Neurol"},{"key":"10.1016\/j.braindev.2026.104545_bb0135","doi-asserted-by":"crossref","first-page":"10287","DOI":"10.3389\/dyst.2022.10287","article-title":"Approach to the treatment of pediatric dystonia","volume":"1","author":"Gorodetsky","year":"2022","journal-title":"Dystonia"},{"key":"10.1016\/j.braindev.2026.104545_bb0140","doi-asserted-by":"crossref","first-page":"37","DOI":"10.1016\/j.neuroscience.2023.02.020","article-title":"Striatal subregion-selective dysregulated dopamine receptor-mediated intracellular signaling in a model of DOPA-responsive dystonia","volume":"517","author":"Roman","year":"2023","journal-title":"Neuroscience"},{"key":"10.1016\/j.braindev.2026.104545_bb0145","doi-asserted-by":"crossref","DOI":"10.1016\/j.intimp.2024.111900","article-title":"Ischemia-inhibited ferric chelate reductase 1 improves ferroptosis-mediated intestinal ischemia injury via hippo signaling","volume":"132","author":"Liang","year":"2024","journal-title":"Int Immunopharmacol"},{"issue":"4","key":"10.1016\/j.braindev.2026.104545_bb0150","doi-asserted-by":"crossref","first-page":"483","DOI":"10.1007\/s00702-020-02290-z","article-title":"Emerging and converging molecular mechanisms in dystonia","volume":"128","author":"Gonzalez-Latapi","year":"2021","journal-title":"J Neural Transm (Vienna)"},{"issue":"11","key":"10.1016\/j.braindev.2026.104545_bb0155","doi-asserted-by":"crossref","first-page":"3328","DOI":"10.1093\/jxb\/erad062","article-title":"Assembly, transfer, and fate of mitochondrial iron-sulfur clusters","volume":"74","author":"Pedroletti","year":"2023","journal-title":"J Exp Bot"},{"issue":"4","key":"10.1016\/j.braindev.2026.104545_bb0160","doi-asserted-by":"crossref","first-page":"502","DOI":"10.1097\/WCO.0000000000001076","article-title":"The apparent paradox of phenotypic diversity and shared mechanisms across dystonia syndromes","volume":"35","author":"Di Fonzo","year":"2022","journal-title":"Curr Opin Neurol"},{"issue":"4","key":"10.1016\/j.braindev.2026.104545_bb0165","doi-asserted-by":"crossref","first-page":"417","DOI":"10.1007\/s00702-020-02269-w","article-title":"Combined dystonias: clinical and genetic updates","volume":"128","author":"Weissbach","year":"2021","journal-title":"J Neural Transm (Vienna)"},{"issue":"9","key":"10.1016\/j.braindev.2026.104545_bb0170","doi-asserted-by":"crossref","first-page":"1038","DOI":"10.1111\/dmcn.14874","article-title":"Pharmacological and neurosurgical interventions for individuals with cerebral palsy and dystonia: a systematic review update and meta-analysis","volume":"63","author":"Bohn","year":"2021","journal-title":"Dev Med Child Neurol"},{"issue":"193","key":"10.1016\/j.braindev.2026.104545_bb0175","doi-asserted-by":"crossref","first-page":"244","DOI":"10.24976\/Discov.Med.202537193.20","article-title":"Channels out of order: a review of central and peripheral nervous system channelopathies","volume":"37","author":"Verriello","year":"2025","journal-title":"Discov Med"},{"issue":"3","key":"10.1016\/j.braindev.2026.104545_bb0180","doi-asserted-by":"crossref","first-page":"223","DOI":"10.1016\/j.pedneo.2024.03.013","article-title":"Insights into clinical phenotypes and treatment responses in a small cohort of Taiwanese patients with SCN1A variants: a preliminary study","volume":"66","author":"Syu","year":"2025","journal-title":"Pediatr Neonatol"}],"container-title":["Brain and Development"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S038776042600046X?httpAccept=text\/xml","content-type":"text\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/api.elsevier.com\/content\/article\/PII:S038776042600046X?httpAccept=text\/plain","content-type":"text\/plain","content-version":"vor","intended-application":"text-mining"}],"deposited":{"date-parts":[[2026,6,1]],"date-time":"2026-06-01T06:48:59Z","timestamp":1780296539000},"score":1,"resource":{"primary":{"URL":"https:\/\/linkinghub.elsevier.com\/retrieve\/pii\/S038776042600046X"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,6]]},"references-count":36,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2026,6]]}},"alternative-id":["S038776042600046X"],"URL":"https:\/\/doi.org\/10.1016\/j.braindev.2026.104545","relation":{},"ISSN":["0387-7604"],"issn-type":[{"value":"0387-7604","type":"print"}],"subject":[],"published":{"date-parts":[[2026,6]]},"assertion":[{"value":"Elsevier","name":"publisher","label":"This article is maintained by"},{"value":"Genotype-dependent response to combined levodopa\/benserazide and trihexyphenidyl in pediatric genetic dystonia","name":"articletitle","label":"Article Title"},{"value":"Brain and Development","name":"journaltitle","label":"Journal Title"},{"value":"https:\/\/doi.org\/10.1016\/j.braindev.2026.104545","name":"articlelink","label":"CrossRef DOI link to publisher maintained version"},{"value":"article","name":"content_type","label":"Content Type"},{"value":"\u00a9 2024 The Japanese Society of Child Neurology. Elsevier B.V. CC BY 4.0","name":"copyright","label":"Copyright"}],"article-number":"104545"}}