Reversible striatal hypermetabolism in chorea associated with moyamoya disease: a report of two cases
Reversible striatal hypermetabolism in chorea associated with moyamoya disease: a report of two cases
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烟雾病相关舞蹈症可逆性纹状体代谢亢进：两例报告
DOI：
10.1007/s00381-016-3111-5
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发表时间：
2016
期刊：
影响因子：
--
通讯作者：
Miyamoto S.
中科院分区：
文献类型：
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作者：
Sugita Y;Funaki T;Takahashi JC;Takagi Y;Fushimi Y;Kikuchi T;Yoshida K;Hatano T;Sasaki N;Miyamoto S.
背景舞蹈病作为小儿烟雾病的表现的病理生理学机制仍然未知，尽管缺血被怀疑是一个可能的原因。作者描述了两例小儿烟雾病病例，均在成功进行搭桥手术后在稳定期出现偏侧舞蹈症。 临床表现 一名病例的脑血流量几乎正常，另一名病例由于术前发生梗塞，导致基底节和分水岭区域的脑血流量减少。在这两种情况下，18F-氟脱氧葡萄糖正电子发射断层扫描显示相应侧纹状体的葡萄糖代谢升高，舞蹈病恢复后恢复正常。磁共振血管造影显示，在代谢亢进病变的确切部位存在扩张和延长的豆纹动脉。
BackgroundThe pathophysiological mechanism of chorea as a presentation of pediatric moyamoya disease remains unknown, although ischemia is suspected as a likely cause. The authors describe two cases of pediatric moyamoya disease, both of which presented with hemichorea in the stable phase after successful bypass surgery.Clinical PresentationCerebral blood flow was almost normal in one case and decreased in the basal ganglia and watershed area in the other case due to infarcts occurring before surgery. In both cases,18F-fluorodeoxyglucose positron emission tomography revealed elevated glucose metabolism in the corresponding side of the striatum, which reverted to normal after recovery from chorea. Magnetic resonance angiography revealed a dilated and extended lenticulostriate artery at the exact site of the hypermetabolic lesion.