Talbot-Stetsko Haley K, Saleh Sara, Brent Ashley, Camelo-Piragua Sandra, Gordon David, Williamson Craig A
School of Medicine, University of Michigan, Ann Arbor, MI, USA.
Department of Neurosurgery, University of Michigan, Ann Arbor, MI, USA.
Neurohospitalist. 2023 Oct;13(4):410-414. doi: 10.1177/19418744231175561. Epub 2023 May 11.
Basilar artery occlusion (BAO) is a rare cause of stroke associated with significant morbidity and mortality. It is most frequently thromboembolic in nature, but may be caused by vertebral artery dissection. We present a case of BAO in a 36-year-old woman with Alport syndrome. She was treated with emergent thrombectomy via the right vertebral artery with return to baseline neurological status. Her clinical status deteriorated later the same day and she was found to have re-occlusion. Repeat thrombectomy was complicated by persistent re-occlusion requiring 7 passes to achieve reperfusion. Unfortunately, her neurological exam remained poor and she was transitioned to comfort care, expiring on admission day 3. An autopsy demonstrated acute dissection of the left vertebral artery, basilar artery, and bilateral posterior cerebral arteries. Alport syndrome is a type IV collagenopathy most known for causing kidney disease. It may also be associated with vascular fragility as type IV collagen forms a significant component of the vascular basement membrane. There are reports of aortic, coronary, and cervical dissections, but few reports of intracranial dissections in patients with Alport syndrome. While iatrogenic dissection cannot be ruled out, the histological findings in this case are most consistent with spontaneous arterial dissection as the cause of her initial neurologic presentation. This highlights the need for further investigation into the relationship between Alport syndrome and vascular fragility and should alert clinicians to the possibility of intracranial dissection in patients with AS.
基底动脉闭塞(BAO)是一种罕见的中风病因,与显著的发病率和死亡率相关。其本质上最常见的是血栓栓塞性的,但也可能由椎动脉夹层引起。我们报告一例患有奥尔波特综合征的36岁女性发生基底动脉闭塞的病例。她接受了经右椎动脉的紧急血栓切除术,神经功能恢复至基线状态。但在同一天晚些时候她的临床状况恶化,发现再次闭塞。重复血栓切除术因持续再闭塞而复杂化,需要进行7次操作才能实现再灌注。不幸的是,她的神经检查结果仍然很差,随后转为舒适护理,于入院第3天死亡。尸检显示左椎动脉、基底动脉和双侧大脑后动脉急性夹层。奥尔波特综合征是一种IV型胶原病,最以导致肾脏疾病而闻名。它也可能与血管脆性有关,因为IV型胶原是血管基底膜的重要组成部分。有关于主动脉、冠状动脉和颈部夹层的报道,但奥尔波特综合征患者颅内夹层的报道很少。虽然不能排除医源性夹层,但该病例的组织学发现最符合自发性动脉夹层是其最初神经症状的原因。这凸显了进一步研究奥尔波特综合征与血管脆性之间关系的必要性,并应提醒临床医生注意患有奥尔波特综合征患者发生颅内夹层的可能性。