{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,12]],"date-time":"2026-05-12T04:43:09Z","timestamp":1778560989199,"version":"3.51.4"},"reference-count":18,"publisher":"Scientific Scholar","content-domain":{"domain":["surgicalneurologyint.com"],"crossmark-restriction":true},"short-container-title":[],"abstract":"<jats:sec id=\"st1\">\n<jats:title>Background: <\/jats:title>\n<jats:p>In this report, we describe rare two pediatric cases that developed oro-mandibular dystonia due to moyamoya disease.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"st2\">\n<jats:title>Case Description: <\/jats:title>\n<jats:p>A 7-year-old boy presented with oro-mandibular dystonia and transient weakness of the left extremities, and was diagnosed as moyamoya disease. Another 7-year-old boy developed oro-mandibular dystonia alone and was diagnosed as moyamoya disease. In both, cerebral blood flow (CBF) was markedly decreased in the involved hemispheres, including the basal ganglia and cerebral cortex. They successfully underwent combined bypass surgery and experienced no further attacks of oromandibular dystonia during follow-up periods. CBF almost normalized through surgical collaterals through direct and indirect bypass.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"st3\">\n<jats:title>Conclusion: <\/jats:title>\n<jats:p>When treating patients with oro-mandibular dystonia, moyamoya disease should be listed as one of the differential diseases. The underlying mechanism of oro-mandibular dystonia in moyamoya disease is still unclear, but persistent cerebral ischemia in the basal ganglia and\/or parietal lobe may play a key role to induce this rare symptom.<\/jats:p>\n<\/jats:sec>","DOI":"10.25259\/sni_661_2021","type":"journal-article","created":{"date-parts":[[2021,9,6]],"date-time":"2021-09-06T07:10:10Z","timestamp":1630912210000},"page":"449","update-policy":"https:\/\/doi.org\/10.25259\/crossmark_policy","source":"Crossref","is-referenced-by-count":3,"title":["Oro-mandibular dystonia in pediatric moyamoya disease: Two cases report"],"prefix":"10.25259","volume":"12","author":[{"given":"Yuichiro","family":"Koga","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Daina","family":"Kashiwazaki","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Emiko","family":"Hori","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Naoki","family":"Akioka","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Satoshi","family":"Kuroda","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"10894","published-online":{"date-parts":[[2021,9,6]]},"reference":[{"key":"10.25259\/SNI_661_2021\/ref-1","doi-asserted-by":"crossref","first-page":"313","DOI":"10.3171\/2012.11.PEDS12199","article-title":"Chorea in the clinical presentation of moyamoya disease: Results of surgical revascularization and a proposed clinicopathological correlation","volume":"11","author":"Ahn","year":"2013","journal-title":"J Neurosurg Pediatr"},{"key":"10.25259\/SNI_661_2021\/ref-2","doi-asserted-by":"crossref","first-page":"262","DOI":"10.5334\/tohm.209","article-title":"Sudden onset of oromandibular dystonia after cerebellar stroke","volume":"4","author":"Akin","year":"2014","journal-title":"Tremor Other Hyperkinet Mov (NY)"},{"key":"10.25259\/SNI_661_2021\/ref-3","doi-asserted-by":"crossref","first-page":"1482","DOI":"10.1002\/mds.23130","article-title":"Movement disorders associated with moyamoya disease: A report of 4 new cases and a review of literatures","volume":"25","author":"Baik","year":"2010","journal-title":"Mov Disord"},{"key":"10.25259\/SNI_661_2021\/ref-4","doi-asserted-by":"crossref","first-page":"1183","DOI":"10.1002\/mds.1213","article-title":"Action hand dystonia after cortical parietal infarction","volume":"16","author":"Burguera","year":"2001","journal-title":"Mov Disord"},{"key":"10.25259\/SNI_661_2021\/ref-5","doi-asserted-by":"crossref","first-page":"1491","DOI":"10.1038\/nn1544","article-title":"The cerebellum communicates with the basal ganglia","volume":"8","author":"Hoshi","year":"2005","journal-title":"Nat Neurosci"},{"key":"10.25259\/SNI_661_2021\/ref-6","doi-asserted-by":"crossref","first-page":"877","DOI":"10.3171\/jns.2004.100.5.0877","article-title":"Involuntary movement induced by cerebral ischemia: Pathogenesis and surgical outcome","volume":"100","author":"Im","year":"2004","journal-title":"J Neurosurg"},{"key":"10.25259\/SNI_661_2021\/ref-7","doi-asserted-by":"crossref","first-page":"1056","DOI":"10.1016\/S1474-4422(08)70240-0","article-title":"Moyamoya disease: Current concepts and future perspectives","volume":"7","author":"Kuroda","year":"2008","journal-title":"Lancet Neurol"},{"key":"10.25259\/SNI_661_2021\/ref-8","doi-asserted-by":"crossref","first-page":"1093","DOI":"10.1227\/01.NEU.0000369606.00861.91","article-title":"Novel bypass surgery for moyamoya disease using pericranial flap: Its impacts on cerebral hemodynamics and long-term outcome","volume":"66","author":"Kuroda","year":"2010","journal-title":"Neurosurgery"},{"key":"10.25259\/SNI_661_2021\/ref-9","doi-asserted-by":"crossref","first-page":"909","DOI":"10.3171\/2019.12.JNS192938","article-title":"Late (5-20 years) outcomes after STA-MCA anastomosis and encephalo-duro-myo-arteriopericranial synangiosis in patients with moyamoya disease","volume":"134","author":"Kuroda","year":"2020","journal-title":"J Neurosurg"},{"key":"10.25259\/SNI_661_2021\/ref-10","doi-asserted-by":"crossref","first-page":"302","DOI":"10.1006\/exnr.1993.1064","article-title":"Cerebellectomy eliminates the motor syndrome of the genetically dystonic rat","volume":"120","author":"LeDoux","year":"1993","journal-title":"Exp Neurol"},{"key":"10.25259\/SNI_661_2021\/ref-11","doi-asserted-by":"crossref","first-page":"885","DOI":"10.1007\/s00381-013-2339-6","article-title":"Involuntary movement in pediatric moyamoya disease patients: Consideration of pathogenetic mechanism using neuroimaging studies","volume":"30","author":"Lee","year":"2014","journal-title":"Childs Nerv Syst"},{"key":"10.25259\/SNI_661_2021\/ref-12","doi-asserted-by":"crossref","first-page":"225","DOI":"10.1016\/j.ejpn.2018.01.005","article-title":"Basal ganglia mechanisms in action selection, plasticity, and dystonia","volume":"22","author":"Mink","year":"2018","journal-title":"Eur J Paediatr Neurol"},{"key":"10.25259\/SNI_661_2021\/ref-13","doi-asserted-by":"crossref","first-page":"185","DOI":"10.1016\/j.nbd.2011.01.026","article-title":"The functional neuroanatomy of dystonia","volume":"42","author":"Neychev","year":"2011","journal-title":"Neurobiol Dis"},{"key":"10.25259\/SNI_661_2021\/ref-14","doi-asserted-by":"crossref","first-page":"559","DOI":"10.3171\/2010.9.PEDS10192","article-title":"Patients with moyamoya disease presenting with movement disorder","volume":"6","author":"Pandey","year":"2010","journal-title":"J Neurosurg Pediatr"},{"key":"10.25259\/SNI_661_2021\/ref-15","first-page":"73","article-title":"Etiology, diagnosis and management of oromandibular dystonia: An update for stomatologists","volume":"18","author":"Raoofi","year":"2017","journal-title":"J Dent (Shiraz)"},{"key":"10.25259\/SNI_661_2021\/ref-16","first-page":"142","article-title":"Long-term outcome in children with moyamoya syndrome after cranial revascularization by pial synangiosis","volume":"100","author":"Scott","year":"2004","journal-title":"J Neurosurg"},{"key":"10.25259\/SNI_661_2021\/ref-17","doi-asserted-by":"crossref","first-page":"2243","DOI":"10.1007\/s00381-016-3111-5","article-title":"Reversible striatal hypermetabolism in chorea associated with moyamoya disease: A report of two cases","volume":"32","author":"Sugita","year":"2016","journal-title":"Childs Nerv Syst"},{"key":"10.25259\/SNI_661_2021\/ref-18","doi-asserted-by":"crossref","first-page":"288","DOI":"10.1001\/archneur.1969.00480090076012","article-title":"Cerebrovascular \u201cmoyamoya\u201d disease. Disease showing abnormal net-like vessels in base of brain","volume":"20","author":"Suzuki","year":"1969","journal-title":"Arch Neurol"}],"container-title":["Surgical Neurology International"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/surgicalneurologyint.com\/oro-mandibular-dystonia-in-pediatric-moyamoya-disease-two-cases-report\/","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/surgicalneurologyint.com\/surgicalint-articles\/oro-mandibular-dystonia-in-pediatric-moyamoya-disease-two-cases-report\/","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,9,6]],"date-time":"2021-09-06T07:10:28Z","timestamp":1630912228000},"score":1,"resource":{"primary":{"URL":"http:\/\/surgicalneurologyint.com\/surgicalint-articles\/oro-mandibular-dystonia-in-pediatric-moyamoya-disease-two-cases-report\/"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,9,6]]},"references-count":18,"alternative-id":["10.25259\/SNI_661_2021"],"URL":"https:\/\/doi.org\/10.25259\/sni_661_2021","relation":{},"ISSN":["2152-7806"],"issn-type":[{"value":"2152-7806","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,9,6]]},"article-number":"449"}}