Moyamoya disease presenting with chorea

Watanabe, Kazuyoshi; Negoro, Tamiko; Maehara, Mitsuo; Takahashi, Izumi; Nomura, Kazushi; Miura, Kiyokuni

doi:10.1016/0887-8994(90)90077-e

Cited by 35 publications

(26 citation statements)

References 2 publications

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“…5,9,11,12,18,[23][24][25] In our series, 6 (60%) of the 10 patients displayed chorea as the initial presenting sign of their disease. Notably, 3 of these patients were given the diagnosis of Sydenham chorea with suspicion of rheumatic fever despite not meeting the Jones criteria.…”

Section: Discussionmentioning

confidence: 53%

“…In some of these reported cases, the chorea resolved after surgical revascularization. 7,11,17,24,25 In the present study, our aim was to characterize those patients with moyamoya disease who presented with chorea in terms of clinical course, radiographic findings, and response to surgical revascularization. In our analysis, we had hypothesized that the presence of hypertrophied collateral vessels in the basal ganglia provides the pathological substrate for the chorea.…”

Section: ©Aans 2013mentioning

confidence: 99%

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Chorea in the clinical presentation of moyamoya disease: results of surgical revascularization and a proposed clinicopathological correlation

Ahn

Scott²,

Robertson

et al. 2013

PED

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Object 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. Methods 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. Results 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 3.8–17.9 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). Conclusions 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.

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Section: Discussionmentioning

confidence: 53%

Section: ©Aans 2013mentioning

confidence: 99%

Chorea in the clinical presentation of moyamoya disease: results of surgical revascularization and a proposed clinicopathological correlation

Ahn

Scott²,

Robertson

et al. 2013

PED

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“…These may include chorea, dystonia, and dyskinesia. [23] MMS however, has rarely been reported to have these manifestations. Myoclonus has not been reported as a presenting feature in either of the conditions, although acute myoclonus can present as a manifestation of cortical stroke in these patients.…”

Section: Discussionmentioning

confidence: 99%

“…There are quite a few case reports in the literature which have described movement disorders as a manifestation of MMD; however these have been rarely reported in cases of MMS. [23]…”

Section: Introductionmentioning

confidence: 99%

Dystonia, myoclonus, and encephalopathy in a single patient: A rare association of moyamoya

et al. 2016

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Moyamoya disease (MMD) is a rare neurovascular disorder which pathologically, is a chronic cerebrovasculopathy. It is characterized by stenosis of the internal carotid artery (ICA) and the main branches within the circle of Willis with consecutive development of collateral vessels. There are a few cases in the literature which have described movement disorders as a manifestation of MMD; however these have been uncommonly reported in cases of moyamoya syndrome (MMS). We present a 10-year-old boy with dystonia, myoclonus and encephalopathy like features. These features in association with moyamoya, are rarely described.

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“…An unusual case of focal PKD involving the muscles supplied by the lower cranial nerves due to an old medullary hemorrhage has been described 7. Moyamoya disease was reported to be responsible for paroxysmal chorea 8. Paroxysmal dyskinesia was reported in association with transient cerebral ischemia 9…”

Section: Discussionmentioning

confidence: 99%

Orthostatic paroxysmal dystonia

et al. 2002

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A 52-year-old man with a history of cerebrovascular disease presented with a 3-year history of paroxysmal hemidystonia precipitated by assuming an upright position after sitting or lying down. MRA showed occlusion of the contralateral internal carotid artery (ICA) and near-total occlusion of the ipsilateral ICA. Subtraction single proton emission computed tomography demonstrated decreased perfusion in the contralateral frontoparietal cortex during the typical dystonic spell. We have coined the term "orthostatic paroxysmal dystonia" for this phenomenon.

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Moyamoya disease presenting with chorea

Cited by 35 publications

References 2 publications

Chorea in the clinical presentation of moyamoya disease: results of surgical revascularization and a proposed clinicopathological correlation

Chorea in the clinical presentation of moyamoya disease: results of surgical revascularization and a proposed clinicopathological correlation

Dystonia, myoclonus, and encephalopathy in a single patient: A rare association of moyamoya

Orthostatic paroxysmal dystonia

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