Coughlin Daniel J, Miller Charles A, Schuette A Jesse
Department of Neurosurgery, Walter Reed National Military Medical Center, Bethesda, Maryland, USA.
Department of Neurosurgery, Walter Reed National Military Medical Center, Bethesda, Maryland, USA.
World Neurosurg. 2017 Aug;104:1049.e1-1049.e6. doi: 10.1016/j.wneu.2017.05.083. Epub 2017 May 23.
This is the first report of the successful treatment of moyamoya disease and an unruptured intracranial aneurysm in a patient with Floating-Harbor syndrome.
A 35-year-old, phenotypically syndromic woman presented with signs and symptoms consistent with ischemic stroke. Magnetic resonance imaging and catheter angiography confirmed diagnosis of moyamoya and a 6-mm basilar apex artery aneurysm (BAA). She underwent right-sided craniotomy for direct and indirect revascularization by means of superficial temporal artery-middle cerebral artery bypass and encephalo-duro-arterio-synangiosis. Three months later, she underwent stent-assisted coiling of the BAA. At 9 months, the patient remains without her preoperative neurological deficits. Interval catheter angiography confirms revascularization of her right hemisphere and obliteration of her BAA.
We present the first case of diagnosis and treatment of moyamoya disease and BAA in a patient with Floating-Harbor syndrome. Due to a paucity of literature on this rare disorder, there has not been an associated link between Floating-Harbor syndrome and cerebrovascular disease. Our report and literature review suggest that these patients may be prone to cerebrovascular disorders and should be followed closely with neurovascular imaging.
本文首次报道成功治疗一名患有漂浮港综合征患者的烟雾病和未破裂颅内动脉瘤。
一名35岁、具有典型综合征表现的女性,出现与缺血性中风相符的体征和症状。磁共振成像和导管血管造影确诊为烟雾病以及一个6毫米的基底动脉尖动脉瘤(BAA)。她接受了右侧开颅手术,通过颞浅动脉 - 大脑中动脉搭桥和脑 - 硬脑膜 - 动脉 - 血管吻合术进行直接和间接血运重建。三个月后,她接受了BAA的支架辅助弹簧圈栓塞术。九个月时,患者术前的神经功能缺损未再出现。期间的导管血管造影证实其右半球血运重建且BAA闭塞。
我们报道了首例患有漂浮港综合征患者的烟雾病和BAA的诊断与治疗。由于关于这种罕见疾病的文献较少,漂浮港综合征与脑血管疾病之间尚无相关联系。我们的报道及文献综述表明,这些患者可能易患脑血管疾病,应通过神经血管成像密切随访。