Okamura Kazuaki, Higuchi Taro, Izumo Tsuyoshi, Takahira Ryotaro, Sadakata Eisaku, Yoshida Michiharu, Yamaguchi Susumu, Morofuji Yoichi, Baba Shiro, Hiu Takeshi, Matsuo Takayuki
J Neurosurg Case Lessons. 2022 Aug 22;4(8). doi: 10.3171/CASE22238.
Pure subarachnoid hemorrhage (SAH) in patients with moyamoya disease is a rare occurrence. Three underlying mechanisms have been described previously, except for ruptured aneurysm of the circle of Willis. Herein, the authors describe a novel mechanism: rupture of a perforator aneurysm in moyamoya disease.
A 51-year-old man experienced sudden onset of severe headache and vomiting. Computed tomography showed diffuse SAH. Digital subtraction angiography (DSA) showed unilateral moyamoya disease without remarkable etiology of SAH. The patient underwent conservative management with antihypertensive agents. The second DSA on day 17 revealed a slow-filling aneurysm emerging from the basilar top perforating artery. The diagnosis of SAH due to unknown origin was changed to ruptured basilar artery perforator aneurysm (BAPA). The third follow-up DSA on day 159 revealed the resolution of BAPA.
In the case of pure SAH, it is crucial to consider the possibility of perforator aneurysms due to hemodynamic stress caused by moyamoya disease. Repeated DSA is essential for detecting the lesion.
烟雾病患者出现单纯蛛网膜下腔出血(SAH)的情况较为罕见。除了 Willis 环动脉瘤破裂外,先前已描述了三种潜在机制。在此,作者描述了一种新机制:烟雾病中穿支动脉瘤破裂。
一名 51 岁男性突发严重头痛和呕吐。计算机断层扫描显示弥漫性 SAH。数字减影血管造影(DSA)显示单侧烟雾病,SAH 无明显病因。患者接受了抗高血压药物的保守治疗。第 17 天的第二次 DSA 显示从基底动脉顶端穿支动脉出现一个充盈缓慢的动脉瘤。病因不明的 SAH 诊断改为基底动脉穿支动脉瘤(BAPA)破裂。第 159 天的第三次随访 DSA 显示 BAPA 消失。
在单纯 SAH 的情况下,考虑烟雾病引起的血流动力学应激导致穿支动脉瘤的可能性至关重要。重复 DSA 对于检测病变至关重要。