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直接低流量型颈内动脉海绵窦瘘的非典型表现:病例报告及文献复习

Atypical Manifestation of Direct Low-Flow Carotid-Cavernous Fistula: Case Report and Review of the Literature.

作者信息

D'Angelo Luca, Paglia Francesco, Caporlingua Alessandro, Sampirisi Luigi, Guidetti Giulio, Santoro Antonio

机构信息

Department of Neurology and Psychiatry, Neurosurgery, "Sapienza" University of Rome, Rome, Italy.

Department of Neurology and Psychiatry, Neurosurgery, "Sapienza" University of Rome, Rome, Italy.

出版信息

World Neurosurg. 2019 May;125:456-460. doi: 10.1016/j.wneu.2019.02.027. Epub 2019 Feb 26.

DOI:10.1016/j.wneu.2019.02.027
PMID:30818073
Abstract

BACKGROUND

Carotid cavernous fistula (CCF) is a rare type of arteriovenous shunt that develops within the cavernous sinus (CS). Direct CCFs entail a direct communication between the cavernous internal carotid artery and the CS and are typically high-flow lesions. Most CCFs drain into the ophthalmic veins (typical venous drainage pattern), leading to the pathognomic ocular clinical triad associated with a CCF. When an obstruction of the typical venous outflow is present, the arterial pressure generated by the fistula is transmitted into the cerebral venous system via the sphenoparietal sinus, which might lead to intracerebral hemorrhage. We present a rare case of posttraumatic, direct, low-flow CCF associated with cerebral hemorrhage, a typical venous drainage pattern, and without ocular symptoms at presentation.

CASE DESCRIPTION

A 76-year-old woman was hospitalized for a posttraumatic frontotemporopolar hemorrhage associated with multiple fractures of the maxillofacial and cranial base skeleton and midline shift >10 mm. On neurologic examination the Glasgow Coma Scale was 8 and right anisocoria was present. Immediate surgical evacuation of the hematoma was performed. Severe arterial bleeding from the anterior third of the middle cranial fossa floor was controlled intraoperatively. Postoperative brain angio-magnetic resonance imaging and digital subtraction angiography showed a direct CCF without theft phenomenon. Ocular symptoms, and ultimately loss of function of the right eye, appeared 2 weeks from surgery. Endovascular treatment of the CCF was attempted attaining partial closure of the shunt using coils.

CONCLUSIONS

Direct low-flow CCFs are exceedingly rare lesions. Five cases have been described in the literature, 4 of which were associated with spontaneous rupture of a cavernous carotid aneurysm while only 1 case was associated with posttraumatic rupture of a cavernous internal carotid artery pseudoaneurysm. In addition, despite our patient having developed an intraparenchymal hemorrhage most probably correlated to the CCF, the latter was associated with a typical venous drainage via the superior ophthalmic vein, which is uncommonly correlated to intraparenchymal bleeding.

摘要

背景

颈动脉海绵窦瘘(CCF)是一种在海绵窦(CS)内形成的罕见动静脉分流类型。直接型CCF是指海绵窦内颈内动脉与海绵窦之间存在直接交通,通常为高流量病变。大多数CCF经眼静脉引流(典型的静脉引流模式),导致与CCF相关的典型眼部临床三联征。当典型静脉流出道受阻时,瘘管产生的动脉压通过蝶顶窦传入脑静脉系统,这可能导致脑出血。我们报告一例罕见的创伤后直接型低流量CCF,伴有脑出血、典型静脉引流模式,且发病时无眼部症状。

病例描述

一名76岁女性因创伤后额颞极出血入院,伴有颌面和颅底骨骼多处骨折及中线移位>10mm。神经系统检查时格拉斯哥昏迷量表评分为8分,右侧瞳孔不等大。立即进行了血肿的手术清除。术中控制了中颅窝底前三分之一的严重动脉出血。术后脑磁共振血管造影和数字减影血管造影显示为直接型CCF,无盗血现象。术后2周出现眼部症状,最终右眼功能丧失。尝试对CCF进行血管内治疗,使用弹簧圈部分封闭分流。

结论

直接型低流量CCF是极其罕见的病变。文献中已描述5例,其中4例与海绵窦段颈动脉瘤自发破裂有关,仅1例与海绵窦内颈内动脉假性动脉瘤创伤后破裂有关。此外,尽管我们的患者发生了很可能与CCF相关的脑实质内出血,但后者与通过眼上静脉的典型静脉引流有关,而这与脑实质内出血的相关性并不常见。

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