烟雾病相关舞蹈症中的可逆性纹状体代谢亢进：两例报告
Reversible striatal hypermetabolism in chorea associated with moyamoya disease: a report of two cases.
作者信息
Sugita Yoshito, Funaki Takeshi, Takahashi Jun C, Takagi Yasushi, Fushimi Yasutaka, Kikuchi Takayuki, Yoshida Kazumichi, Hatano Taketo, Sasaki Natsuhi, Miyamoto Susumu
机构信息
Department of Neurosurgery, Kyoto University Graduate School of Medicine, 54 Kawahara-cho, Shogoin, Sakyo-ku, Kyoto, 606-8507, Japan.
Department of Neurosurgery, National Cerebral and Cardiovascular Center, Suita, Osaka, Japan.
出版信息
Childs Nerv Syst. 2016 Nov;32(11):2243-2247. doi: 10.1007/s00381-016-3111-5. Epub 2016 May 18.
BACKGROUND
The 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 PRESENTATION
Cerebral 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, F-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.
背景
尽管怀疑缺血是小儿烟雾病出现舞蹈症的可能原因，但其病理生理机制仍不清楚。作者描述了两例小儿烟雾病病例，两例均在成功搭桥手术后的稳定期出现偏侧舞蹈症。
临床表现
一例患者脑血流量基本正常，另一例患者由于术前发生梗死，基底节和分水岭区脑血流量减少。在这两例患者中，氟脱氧葡萄糖正电子发射断层扫描显示纹状体相应侧葡萄糖代谢升高，舞蹈症恢复后恢复正常。磁共振血管造影显示在代谢亢进病变的确切部位，豆纹动脉扩张并延长。
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