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球孢子菌性脑膜炎所致多发性颅内动脉瘤:以动脉瘤形成和自发血栓形成为特征的病例报告及文献复习

Multiple Intracranial Aneurysms from Coccidioidal Meningitis: Case Report Featuring Aneurysm Formation and Spontaneous Thrombosis with Literature Review.

作者信息

Buchanan Ian A, Ravina Kristine, Strickland Ben, Fredrickson Vance, She Rosemary, Mathew Anna, Rennert Robert, Russin Jonathan J

机构信息

Department of Neurological Surgery, Keck School of Medicine, University of Southern California, Los Angeles, USA.

Department of Neurological Surgery, Keck School of Medicine, University of Southern California, Los Angeles, USA.

出版信息

World Neurosurg. 2019 Jan;121:117-123. doi: 10.1016/j.wneu.2018.08.220. Epub 2018 Sep 7.

DOI:10.1016/j.wneu.2018.08.220
PMID:30201579
Abstract

BACKGROUND

Coccidioidal meningitis can progress to vasculitis with aneurysm formation. Although aneurysmogenesis is rare, it carries exceptionally high mortality. Except in one instance, prior case reports have documented universally fatal consequences.

CASE DESCRIPTION

A 26-year-old man developed disseminated coccidioidomycosis with formation of multiple aneurysms throughout the anterior intracranial vasculature bilaterally. This report is unique in that it chronicles the formation and subsequent spontaneous thrombosis of several aneurysms over a 4-week period. In total 10 aneurysms were documented in the same patient-the highest reported to date. The patient was eventually discharged from the hospital for what has heretofore been a universally fatal disease process. Neurologic examination and vascular imaging 1 month after discharge demonstrated stable findings.

CONCLUSIONS

Coccidioidal aneurysms carry a high mortality. The mainstay of therapy remains lifelong triazole antifungal therapy with the addition of liposomal amphotericin in cases of treatment failure. Steroid use is controversial but should be considered whenever there is vascular involvement. Although watchful waiting is reasonable in light of the possibility of spontaneous thrombosis with medical management, dynamic changes in aneurysm size or configuration should prompt timely endovascular or operative interventions.

摘要

背景

球孢子菌性脑膜炎可进展为伴有动脉瘤形成的血管炎。尽管动脉瘤形成罕见,但死亡率极高。除了1例之外,既往病例报告均记录了普遍致命的后果。

病例描述

一名26岁男性患播散性球孢子菌病,双侧颅内前循环血管系统多处形成动脉瘤。本报告的独特之处在于,它记录了数处动脉瘤在4周内的形成及随后的自发血栓形成过程。该患者共记录到10处动脉瘤,为迄今为止报告数量最多的。该患者最终从医院出院,而此前这一疾病过程通常是致命的。出院1个月后的神经学检查和血管成像显示结果稳定。

结论

球孢子菌性动脉瘤死亡率高。治疗的主要方法仍是终身使用三唑类抗真菌药物,治疗失败时加用脂质体两性霉素。类固醇的使用存在争议,但血管受累时应考虑使用。鉴于药物治疗可能出现自发血栓形成,密切观察等待是合理的,但动脉瘤大小或形态的动态变化应促使及时进行血管内或手术干预。

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