Alport综合征患者的椎基底动脉夹层。

IF 0.9 Q4 CLINICAL NEUROLOGY Neurohospitalist Pub Date : 2023-10-01 Epub Date: 2023-05-11 DOI:10.1177/19418744231175561
Haley K Talbot-Stetsko, Sara Saleh, Ashley Brent, Sandra Camelo-Piragua, David Gordon, Craig A Williamson
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引用次数: 0

摘要

基底动脉闭塞(BAO)是一种罕见的中风原因,具有显著的发病率和死亡率。它在自然界中最常见的是血栓栓塞,但可能是由椎动脉夹层引起的。我们报告了一例36岁患有Alport综合征的女性BAO。她接受了经右椎动脉紧急血栓切除术的治疗,恢复了基线神经状态。同一天晚些时候,她的临床状况恶化,被发现再次闭塞。重复血栓切除术因持续再闭塞而变得复杂,需要7次才能实现再灌注。不幸的是,她的神经系统检查仍然很差,她被转移到舒适护理,在入院第3天到期。尸检显示左侧椎动脉、基底动脉和双侧大脑后动脉有急性夹层。Alport综合征是一种IV型胶原病,最常见的是引起肾脏疾病。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。有主动脉、冠状动脉和颈部夹层的报道,但很少有Alport综合征患者颅内夹层的报道。虽然不能排除医源性夹层,但本例的组织学表现与她最初神经系统表现的自发性动脉夹层最为一致。这突出了进一步研究Alport综合征与血管脆性之间关系的必要性,并应提醒临床医生AS患者颅内夹层的可能性。
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Vertebral and Basilar Artery Dissection in a Patient With Alport Syndrome.

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.

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来源期刊
Neurohospitalist
Neurohospitalist CLINICAL NEUROLOGY-
CiteScore
1.60
自引率
0.00%
发文量
108
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