Lazzaro Marc A, Cochran Elizabeth J, Lopes Demetrius K, Prabhakaran Shyam
Department of Neurology, Medical College of Wisconsin and Froedtert Hospital, Milwaukee, WI.
Neurol Int. 2011 Jun;3(1):e3. doi: 10.4081/ni.2011.e3. Epub 2011 Apr 28.
Moyamoya syndrome is a rare cerebrovascular disorder characterized by progressive occlusion of the supraclinoid internal carotid artery and proximal portions of the anterior and middle cerebral arteries resulting in an extensive network of collateralized blood vessels and producing a characteristic angiographic appearance. Although the pathophysiology is unclear, hematologic disorders have been associated with development of the moyamoya syndrome. A case report is presented. A 29 year-old female with a history of essential thrombocythemia developed progressive ischemic strokes. Angiography revealed characteristic moyamoya changes and pathologic examination showed intimal hyperplasia with scant collagen fibers and myxoid change. This is the first reported case of moyamoya syndrome in an adult patient with essential thrombocythemia demonstrating histological findings that suggest a shared pathophysiology with moyamoya syndrome in sickle cell anemia.
烟雾综合征是一种罕见的脑血管疾病,其特征为鞍上颈内动脉以及大脑前动脉和大脑中动脉近端部分进行性闭塞,导致广泛的侧支血管网络形成,并产生特征性的血管造影表现。尽管其病理生理学尚不清楚,但血液系统疾病已被认为与烟雾综合征的发生有关。本文报告一例病例。一名29岁患有原发性血小板增多症的女性出现了进行性缺血性中风。血管造影显示有典型的烟雾病改变,病理检查显示内膜增生,胶原纤维稀少且有黏液样改变。这是首例报道的患有原发性血小板增多症的成年烟雾综合征患者,其组织学表现提示与镰状细胞贫血的烟雾综合征有共同的病理生理学机制。