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題名 | Moyamoya Syndrome with Inherited Proteins C and S Deficiency: Report of One Case=毛毛樣腦血管症候群合併遺傳性C及S蛋白缺乏症:一病例報告 |
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作者姓名(中文) | 張寶玲; 李旺祚; 廖漢文; 林凱信; | 書刊名 | 臺灣兒科醫學會雜誌 |
卷期 | 46:1 民94.01-02 |
頁次 | 頁31-34+54 |
分類號 | 415.92 |
關鍵詞 | 毛毛樣腦血管症; C及S蛋白之缺乏; 缺血性腦中風; Moyamoya syndrome; Proteins C and S deficiencies; Ischemic stroke; |
語文 | 英文(English) |
英文摘要 | Moyamoya disease (MMD) is a vascular abnormality characterized by progressive narrowing of the internal carotid, middle, anterior and posterior cerebral arteries and the development of basal collaterals forming the classic angiographic appearance-moyamoya. Protein C (PC) and its cofactor, protein S (PS), which are vitamin-K dependent, act as inhibitors of coagulation cascade by inactivating factor Va and factor Villa and facilitating thrombolysis. Deficiencies of these proteins leading to a prothrombotic state increase the risk of ischemic cerebrovascular accident. Herein we report a 3-year-old girl, who had an acute onset of right-side hemiparesis during an episode of herpetic gingivostomatitis. She was noted to have moyamoya syndrome in the angiographic examination and was later proved to have inherited deficiencies of both PC and PS. She received warfarin medical treatment and then bilateral encephalo-duro-arterio-synangiosis (EDAS) and encephalo-myo-synangiosis (EMS) due to neurological deterioration and is now under the use of low molecular weight heparin for preventing further ischemic stroke. The motor deterioration improved after medical and surgical intervention. We conclude that coagulopathy like PC or PS deficiency should be considered in children presenting with moyamoya syndrome. |
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