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一名儿科患者的新发动静脉畸形:病例报告及文献综述

De Novo Arteriovenous Malformation in a Pediatric Patient: Case Report and Review of the Literature.

作者信息

Santos Roberta, Aguilar-Salinas Pedro, Entwistle John J, Aldana Philipp R, Beier Alexandra D, Hanel Ricardo A

机构信息

Lyerly Neurosurgery, Baptist Health, Jacksonville, Florida, USA.

Division of Pediatric Neurosurgery, University of Florida Health Jacksonville, Jacksonville, Florida, USA.

出版信息

World Neurosurg. 2018 Mar;111:341-345. doi: 10.1016/j.wneu.2017.12.145. Epub 2017 Dec 30.

Abstract

BACKGROUND

An arteriovenous malformation (AVM) consists of a pathologic arteriovenous shunt formed from a tangle of vessels lacking a capillary bed. AVMs were previously accepted as congenital in nature; however, an increase in the number of reported de novo cerebral AVMs challenges the assertion that all AVMs develop in utero hence, the possibility of these lesions presenting postnatally cannot be excluded. A review of literature revealed 31 published cases of de novo AVM formation between 1996 and 2017.

CASE DESCRIPTION

We present the case of a 7-year-old boy with a history of an intracranial aneurysm that was found to have an asymptomatic de novo AVM formation on a routine follow-up angiography. He was being followed since the age of 8 months when the aneurysm was incidentally found during work-up for mild hemiparesis. At that time, magnetic resonance imaging revealed a 4-mm right posterior communicating artery aneurysm along with a right hemispheric stroke and cortical dysplasia. No arteriovenous malformation was noted.

CONCLUSIONS

The exact pathophysiology of de novo AVMs is not completely understood, but a combination of genetic, molecular, and environmental factors may play a role. Our case report only adds to the growing body of clinical evidence supporting the notion that these lesions may be acquired. It would be appropriate to consider an AVM as an abnormal vascular response to an injury rather than a static congenital lesion.

摘要

背景

动静脉畸形(AVM)由一团缺乏毛细血管床的血管形成的病理性动静脉分流组成。AVM以前被认为本质上是先天性的;然而,新报道的脑AVM数量的增加对所有AVM都在子宫内发育这一论断提出了挑战,因此,不能排除这些病变在出生后出现的可能性。对文献的回顾显示,1996年至2017年间共发表了31例新发AVM形成的病例。

病例描述

我们报告一例7岁男孩,有颅内动脉瘤病史,在常规随访血管造影中发现有无症状的新发AVM形成。他自8个月大时起接受随访,当时在对轻度偏瘫进行检查时偶然发现了动脉瘤。当时,磁共振成像显示一个4毫米的右侧后交通动脉瘤,伴有右侧半球中风和皮质发育异常。未发现动静脉畸形。

结论

新发AVM的确切病理生理学尚未完全了解,但遗传、分子和环境因素的综合作用可能起一定作用。我们的病例报告只是增加了越来越多的临床证据,支持这些病变可能是后天获得的这一观点。将AVM视为对损伤的异常血管反应而非静态先天性病变是合适的。

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