{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,12]],"date-time":"2026-05-12T04:43:10Z","timestamp":1778560990053,"version":"3.51.4"},"reference-count":24,"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>Dystonia is a rare movement disorder with some cases being difficult to treat. Although dystonia can occur as a symptom of moyamoya disease, few studies have reported truncal dystonia occurring with middle cerebral artery (MCA) stenosis. Here, we report a case of truncal dystonia with MCA occlusion.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"st2\">\n<jats:title>Case Description: <\/jats:title>\n<jats:p>The patient was a 48-year-old female clerical worker who lived alone. An abnormal cervical posture initially appeared 7 years before (right flexion). Symptoms improved with medication and botulinum toxin injection. Five years before this report, her symptoms worsened, so the dose of oral medication was increased and botulinum treatment was performed, but the symptoms did not improve. The patient showed decreased cerebral blood flow (CBF) in the cortical areas but not in the basal ganglia. We performed superficial temporal artery-MCA bypass surgery because we believed that the dystonia was due to right MCA stenosis. The patient\u2019s symptoms improved immediately after surgery, except for her mild cervical backbend. Seven months after the surgery, the patient\u2019s involuntary movements showed further improvement, and symptoms have not worsened even after 2 years.<\/jats:p>\n<\/jats:sec>\n<jats:sec id=\"st3\">\n<jats:title>Conclusion: <\/jats:title>\n<jats:p>Revascularization therapy improved CBF and truncal dystonia and could be a viable treatment option for dystonia with ischemia in the MCA region. Extensive cerebral ischemia can result in cortical inhibition loss or over-adapted cerebral plasticity and cause dystonia. Revascularization therapy may be useful for patients with dystonia and decreased CBF in the MCA region.<\/jats:p>\n<\/jats:sec>","DOI":"10.25259\/sni_173_2022","type":"journal-article","created":{"date-parts":[[2022,4,15]],"date-time":"2022-04-15T11:25:17Z","timestamp":1650021917000},"page":"155","update-policy":"https:\/\/doi.org\/10.25259\/crossmark_policy","source":"Crossref","is-referenced-by-count":2,"title":["Truncal dystonia with isolated middle cerebral artery ischemia: A case report of revascularization therapy for dystonia"],"prefix":"10.25259","volume":"13","author":[{"given":"Shinichi","family":"Matsumoto","sequence":"first","affiliation":[{"name":"Department of Neurology, Osaka Neurological Institute,"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yuki","family":"Yamamoto","sequence":"additional","affiliation":[{"name":"Department of Clinical Neuroscience, Tokushima University,"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Koji","family":"Fujita","sequence":"additional","affiliation":[{"name":"Department of Clinical Neuroscience, Tokushima University,"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ryosuke","family":"Miyamoto","sequence":"additional","affiliation":[{"name":"Department of Clinical Neuroscience, Tokushima University,"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hidetaka","family":"Koizumi","sequence":"additional","affiliation":[{"name":"Department of Neurology, Osaka Neurological Institute,"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Akihiro","family":"Tateishi","sequence":"additional","affiliation":[{"name":"Departments of Neurosurgery, Osaka Neurological Institute, Toyonaka, Japan,"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Naoaki","family":"Yamada","sequence":"additional","affiliation":[{"name":"Neuroradiology, Osaka Neurological Institute, Toyonaka, Japan."}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Yuishin","family":"Izumi","sequence":"additional","affiliation":[{"name":"Department of Clinical Neuroscience, Tokushima University,"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"10894","published-online":{"date-parts":[[2022,4,15]]},"reference":[{"key":"10.25259\/SNI_173_2022\/ref-1","doi-asserted-by":"crossref","first-page":"981","DOI":"10.1093\/brain\/106.4.981","article-title":"Stereotaxic thalamotomy in 55 cases of dystonia","volume":"106","author":"Andrew","year":"1983","journal-title":"Brain"},{"key":"10.25259\/SNI_173_2022\/ref-2","doi-asserted-by":"crossref","first-page":"13","DOI":"10.14802\/jmd.13003","article-title":"A case of isolated middle cerebral artery stenosis with hemichorea and moyamoya pattern collateralization","volume":"6","author":"Chung","year":"2013","journal-title":"J Mov Disord"},{"key":"10.25259\/SNI_173_2022\/ref-3","doi-asserted-by":"crossref","first-page":"483","DOI":"10.1016\/j.neurol.2016.07.006","article-title":"Movement disorders and stroke","volume":"172","author":"Defebvre","year":"2016","journal-title":"Rev Neurol"},{"key":"10.25259\/SNI_173_2022\/ref-4","doi-asserted-by":"crossref","first-page":"1029","DOI":"10.1002\/acn3.250","article-title":"Cervical dystonia caused by focal putaminal ischemia","volume":"2","author":"Dinkelbach","year":"2015","journal-title":"Ann Clin Transl Neurol"},{"key":"10.25259\/SNI_173_2022\/ref-5","doi-asserted-by":"crossref","first-page":"1017","DOI":"10.1136\/jnnp.2005.087148","article-title":"Bilateral grey-matter increase in the putamen in primary blepharospasm","volume":"77","author":"Etgen","year":"2006","journal-title":"J Neurol Neurosurg Psychiatry"},{"key":"10.25259\/SNI_173_2022\/ref-6","doi-asserted-by":"crossref","first-page":"1","DOI":"10.1212\/WNL.50.5_Suppl_5.S1","article-title":"Classification of dystonia","volume":"78","author":"Fahn","year":"1998","journal-title":"Adv Neurol"},{"key":"10.25259\/SNI_173_2022\/ref-7","doi-asserted-by":"crossref","first-page":"211","DOI":"10.1002\/mds.870030305","article-title":"Pathological report of four patients presenting with cranial dystonias","volume":"3","author":"Gibb","year":"1988","journal-title":"Mov Disord"},{"key":"10.25259\/SNI_173_2022\/ref-8","doi-asserted-by":"crossref","first-page":"646","DOI":"10.1177\/0883073815609152","article-title":"Pial synangiosis ameliorates movement disorders in the absence of prior stroke in moyamoya disease","volume":"31","author":"Greene","year":"2016","journal-title":"J Child Neurol"},{"key":"10.25259\/SNI_173_2022\/ref-9","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_173_2022\/ref-10","doi-asserted-by":"crossref","first-page":"434","DOI":"10.2335\/scs.30.434","article-title":"Japanese EC-IC bypass trial (JET Study): The second interim analysis","volume":"30","author":"JET Study Group","year":"2002","journal-title":"Surg Cereb Stroke"},{"key":"10.25259\/SNI_173_2022\/ref-11","doi-asserted-by":"crossref","first-page":"1740","DOI":"10.1212\/01.wnl.0000246112.19504.61","article-title":"A new twist on the anatomy of dystonia: The basal ganglia and the cerebellum?","volume":"67","author":"Jinnah","year":"2006","journal-title":"Neurology"},{"key":"10.25259\/SNI_173_2022\/ref-12","doi-asserted-by":"crossref","first-page":"390","DOI":"10.2176\/nmc.48.390","article-title":"Dramatic disappearance of moyamoya disease-induced chorea after indirect bypass surgery","volume":"48","author":"Kamijo","year":"2008","journal-title":"Neurol Med Chirurg"},{"key":"10.25259\/SNI_173_2022\/ref-13","doi-asserted-by":"crossref","first-page":"115","DOI":"10.4103\/1817-1745.187629","article-title":"Dystonia an unusual presentation in pediatric Moyamoya disease: Imaging findings of a case","volume":"11","author":"Kumar","year":"2016","journal-title":"J Pediatr Neurosci"},{"key":"10.25259\/SNI_173_2022\/ref-14","doi-asserted-by":"crossref","first-page":"1196","DOI":"10.1002\/mds.10480","article-title":"Epidemiology of primary dystonias in Japan comparison with Western countries","volume":"18","author":"Matsumoto","year":"2003","journal-title":"Mov Disord"},{"key":"10.25259\/SNI_173_2022\/ref-15","doi-asserted-by":"crossref","first-page":"597","DOI":"10.1016\/S1474-4422(13)70057-7","article-title":"Movement disorders in cerebrovascular disease","volume":"12","author":"Mehanna","year":"2013","journal-title":"Lancet Neurol"},{"key":"10.25259\/SNI_173_2022\/ref-16","doi-asserted-by":"crossref","first-page":"693","DOI":"10.1002\/mds.870130415","article-title":"Pallidotomy for generalized dystonia","volume":"13","author":"Ondo","year":"1998","journal-title":"Mov Disord"},{"key":"10.25259\/SNI_173_2022\/ref-17","doi-asserted-by":"crossref","first-page":"2871","DOI":"10.1093\/brain\/awp213","article-title":"Abnormal sensorimotor plasticity in organic but not in psychogenic dystonia","volume":"132","author":"Quartarone","year":"2009","journal-title":"Brain"},{"key":"10.25259\/SNI_173_2022\/ref-18","first-page":"e17272","article-title":"Dystonia as a presenting feature of acute ischemic stroke: A case report and literature review","volume":"13","author":"Roman Casul","year":"2021","journal-title":"Cureus"},{"key":"10.25259\/SNI_173_2022\/ref-19","doi-asserted-by":"crossref","first-page":"829","DOI":"10.5692\/clinicalneurol.cn-001350","article-title":"Clinical findings of a patient with hemiballism after superficial temporal artery-middle cerebral artery anastomosis for idiopathic middle cerebral artery stenosis","volume":"59","author":"Shibata","year":"2019","journal-title":"Rinsho Shinkeigaku"},{"key":"10.25259\/SNI_173_2022\/ref-20","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_173_2022\/ref-21","doi-asserted-by":"crossref","first-page":"14","DOI":"10.1186\/s40348-015-0026-2","article-title":"Post stroke hemidystonia in children: A neglected area of research","volume":"2","author":"Tibussek","year":"2015","journal-title":"Mol Cell Pediatr"},{"key":"10.25259\/SNI_173_2022\/ref-22","doi-asserted-by":"crossref","first-page":"459","DOI":"10.1056\/NEJMoa042187","article-title":"Bilateral deep-brain stimulation of the globus pallidus in primary generalized dystonia","volume":"352","author":"Vidailhet","year":"2005","journal-title":"N Engl J Med"},{"key":"10.25259\/SNI_173_2022\/ref-23","doi-asserted-by":"crossref","first-page":"274","DOI":"10.1007\/s00381-004-1104-2","article-title":"A case of moyamoya disease presenting with chorea","volume":"22","author":"Zheng","year":"2006","journal-title":"Childs Nerv Syst"},{"key":"10.25259\/SNI_173_2022\/ref-24","doi-asserted-by":"crossref","first-page":"702","DOI":"10.1212\/WNL.38.5.702","article-title":"Pathology in brainstem regions of individuals with primary dystonia","volume":"38","author":"Zweig","year":"1988","journal-title":"Neurology"}],"container-title":["Surgical Neurology International"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/surgicalneurologyint.com\/truncal-dystonia-with-isolated-middle-cerebral-artery-ischemia-a-case-report-of-revascularization-therapy-for-dystonia\/","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"http:\/\/surgicalneurologyint.com\/surgicalint-articles\/truncal-dystonia-with-isolated-middle-cerebral-artery-ischemia-a-case-report-of-revascularization-therapy-for-dystonia\/","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2022,4,15]],"date-time":"2022-04-15T11:25:36Z","timestamp":1650021936000},"score":1,"resource":{"primary":{"URL":"http:\/\/surgicalneurologyint.com\/surgicalint-articles\/truncal-dystonia-with-isolated-middle-cerebral-artery-ischemia-a-case-report-of-revascularization-therapy-for-dystonia\/"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,4,15]]},"references-count":24,"alternative-id":["10.25259\/SNI_173_2022"],"URL":"https:\/\/doi.org\/10.25259\/sni_173_2022","relation":{},"ISSN":["2152-7806"],"issn-type":[{"value":"2152-7806","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,4,15]]},"article-number":"155"}}