{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T04:02:09Z","timestamp":1787025729297,"version":"build-2736575974"},"reference-count":25,"publisher":"Journal of Neurosurgery Publishing Group (JNSPG)","issue":"3","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["PED"],"published-print":{"date-parts":[[2013,3]]},"abstract":"<jats:sec>\n                    <jats:title>Object<\/jats:title>\n                    <jats:p>Chorea is a movement disorder characterized by brief, irregular, involuntary contractions that appear to flow from 1 muscle to another. There are a limited number of reports in the literature that have linked moyamoya disease and chorea. The authors describe their experience in treating moyamoya disease in patients in whom chorea developed as part of the clinical presentation.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>The authors conducted a retrospective review of a consecutive series of 316 children who underwent pial synangiosis revascularization for moyamoya disease at the Boston Children's Hospital.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Of 316 surgically treated patients with moyamoya disease, 10 (3.2%; 6 boys and 4 girls) had chorea as a part of their presentation. The average age at surgical treatment was 9.9 years (range 3.8\u201317.9 years). All patients had evidence of hypertrophied lenticulostriate collateral vessels through the basal ganglia on preoperative angiography and\/or MRI on affected sides. Two patients had cystic lesions in the basal ganglia. Nine patients underwent bilateral craniotomies for pial synangiosis, and 1 patient underwent a single craniotomy for unilateral disease. Follow-up was available in 9 patients (average 50.1 months). The mean duration of chorea was 1.36 years (range 2 days to 4 years), with resolution of symptoms in all patients. One patient developed chorea 3 years after surgical treatment, 4 patients had transient chorea that resolved prior to surgery, and 5 patients experienced resolution of the chorea after surgery (average 13 months).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>The authors describe children with moyamoya disease and chorea as part of their clinical presentation. The data suggest that involvement of the basal ganglia by the hypertrophied collateral vessels contributes to the development of chorea, which can wax or wane depending on disease stage or involution of the vessels after revascularization surgery. In most patients, however, the chorea improves or disappears about 1 year after presentation.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.3171\/2012.11.peds12199","type":"journal-article","created":{"date-parts":[[2013,1,4]],"date-time":"2013-01-04T10:01:07Z","timestamp":1357293667000},"page":"313-319","source":"Crossref","is-referenced-by-count":37,"title":["Chorea in the clinical presentation of moyamoya disease: results of surgical revascularization and a proposed clinicopathological correlation"],"prefix":"10.3171","volume":"11","author":[{"given":"Edward S.","family":"Ahn","sequence":"first","affiliation":[{"name":"Division of Pediatric Neurosurgery, Johns Hopkins University School of Medicine, Baltimore, Maryland; and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"R. Michael","family":"Scott","sequence":"additional","affiliation":[{"name":"Departments of Neurosurgery and"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Richard L.","family":"Robertson","sequence":"additional","affiliation":[{"name":"Radiology, Boston Children's Hospital, Harvard Medical School, Boston, Massachusetts"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Edward R.","family":"Smith","sequence":"additional","affiliation":[{"name":"Departments of Neurosurgery and"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1754","reference":[{"key":"b1-peds12199","series-title":"Pediatr Neurosurg","doi-asserted-by":"publisher","first-page":"26","DOI":"10.1159\/000120932","article-title":"Pial synangiosis for moyamoya syndrome in children","volume":"23","author":"Adelson","year":"1995"},{"key":"b2-peds12199","series-title":"Brain","doi-asserted-by":"publisher","first-page":"859","DOI":"10.1093\/brain\/117.4.859","article-title":"The behavioural and motor consequences of focal lesions of the basal ganglia in man","volume":"117","author":"Bhatia","year":"1994"},{"key":"b3-peds12199","series-title":"Mov Disord","doi-asserted-by":"publisher","first-page":"1051","DOI":"10.1002\/mds.10483","article-title":"Moyamoya-induced paroxysmal dyskinesia","volume":"18","author":"Gonzalez-Alegre","year":"2003"},{"key":"b4-peds12199","series-title":"No To Hattatsu","first-page":"166","article-title":"[A case of unilateral moyamoya disease presenting with hemichorea.]","volume":"33","author":"Hama","year":"2001"},{"key":"b5-peds12199","series-title":"J Neurol Neurosurg Psychiatry","doi-asserted-by":"publisher","first-page":"833","DOI":"10.1136\/jnnp.69.6.833","article-title":"Moyamoya disease presenting with singing induced chorea","volume":"69","author":"Han","year":"2000"},{"key":"b6-peds12199","series-title":"Annual Report of Research Group on the Occlusive Disease of the Circle of Willis","first-page":"14","article-title":"Filing of 1500 cases of the occlusive disease of the circle of Willis: appendix, a follow-up study of 5 years or more on 200 cases","author":"Handa","year":"1985"},{"key":"b7-peds12199","series-title":"Mov Disord","doi-asserted-by":"publisher","first-page":"1380","DOI":"10.1002\/mds.10245","article-title":"Hemichorea as an initial manifestation of moyamoya disease: reversible striatal hypoperfusion demonstrated on single photon emission computed tomography","volume":"17","author":"Hong","year":"2002"},{"key":"b8-peds12199","series-title":"Childs Nerv Syst","doi-asserted-by":"publisher","first-page":"1297","DOI":"10.1007\/s00381-010-1209-8","article-title":"Moyamoya disease in children","volume":"26","author":"Ibrahimi","year":"2010"},{"key":"b9-peds12199","series-title":"Arch Neurol","first-page":"1510","article-title":"Hemidystonia and hemichoreoathetosis as an initial manifestation of moyamoya disease","volume":"57","author":"Lyoo","year":"2000"},{"key":"b10-peds12199","series-title":"Neurosurgery","doi-asserted-by":"publisher","first-page":"401","DOI":"10.1227\/00006123-199209000-00003","article-title":"Surgical treatment of moyamoya disease in pediatric patients\u2014 comparison between the results of indirect and direct revascularization procedures","volume":"31","author":"Matsushima","year":"1992"},{"key":"b11-peds12199","series-title":"J Neurosurg Pediatr","doi-asserted-by":"publisher","first-page":"559","DOI":"10.3171\/2010.9.PEDS10192","article-title":"Patients with moyamoya disease presenting with movement disorder. Report of 4 cases","volume":"6","author":"Pandey","year":"2010"},{"key":"b12-peds12199","series-title":"Indian Pediatr","first-page":"1005","article-title":"Chorea: an unusual presenting feature in pediatric Moyamoya disease","volume":"37","author":"Parmar","year":"2000"},{"key":"b13-peds12199","series-title":"Mov Disord","doi-asserted-by":"publisher","first-page":"347","DOI":"10.1002\/mds.870060414","article-title":"Steroid-responsive chorea in moyamoya disease","volume":"6","author":"Pavlakis","year":"1991"},{"key":"b14-peds12199","series-title":"Rev Neurol (Paris)","first-page":"393","article-title":"[Angiodysplasia of moya-moya type disclosed by choreic unvoluntary abnormal movements during oral contraception. 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