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颅内硬脑膜动静脉瘘导致的复发性丘脑症状:罕见病例报告及治疗结果

Recurring Thalamic Symptoms Due to Intracranial Dural Arteriovenous Fistula: Report of Unusual Case and Treatment Outcome.

作者信息

Vinayagamani Selvadasan, Kannath Santhosh Kumar, Rajan Jayadevan Enakshy

机构信息

Department of Imaging Sciences and Interventional Radiology, Sree Chitra Institute for Medical Sciences and Technology, Kerala, India.

Neurointervention Center, Department of Imaging Sciences and Interventional Radiology, Sree Chitra Institute for Medical Sciences and Technology, Kerala, India.

出版信息

World Neurosurg. 2018 Oct;118:9-13. doi: 10.1016/j.wneu.2018.06.187. Epub 2018 Jun 30.

DOI:10.1016/j.wneu.2018.06.187
PMID:29969738
Abstract

BACKGROUND

Initial clinical presentation of dural arteriovenous fistula (DAVF) with predominantly thalamic symptoms is rare and has not been reported until now.

CASE DESCRIPTION

A young child presenting with complaints of tinnitus and mild right hemiparesis was evaluated with an initial magnetic resonance imaging, which revealed a Borden type 2 DAVF in the right transverse sinus with retrograde venous drainage and cortical venous reflux. Flow-related small aneurysms were also noted in the left basal vein of Rosenthal (BVR). Two days later, his motor power deteriorated and he developed right hemisensory loss and severe thalamic pain. Aphasia was atypical and mimicked the transcortical type. Repeat imaging revealed expanding thrombosed aneurysm of BVR with mass effect and edema on thalamocapsular region. Initial antiedema measures reversed the neurologic deficits; however, they recurred, necessitating an urgent endovascular intervention. Angiogram revealed an extensive high-flow DAVF in the right transverse-sigmoid sinus and torcula with preferential retrograde venous drainage into deep veins and left BVR. He underwent staged embolization of DAVF, resulting in significant obliteration of shunt. Neurologic deficits improved in the postoperative period, and the patient remained clinically asymptomatic at 6 months of follow-up.

CONCLUSIONS

DAVF presenting with recurrent predominantly thalamic symptoms is rare. Atypical transcortical aphasia rather than fluent aphasia is an unusual clinical manifestation of thalamic pathology and could result from the involvement of specific thalamic nuclei. Preferential high flow into BVR in the presence of venous anomalies could potentially induce architectural weakness of the venous wall and precipitate aneurysm formation. Embolization of the DAVF can potentially reverse this unusual neurologic condition.

摘要

背景

以丘脑症状为主的硬脑膜动静脉瘘(DAVF)的初始临床表现罕见,迄今为止尚未见报道。

病例描述

一名主诉耳鸣和轻度右侧偏瘫的幼儿接受了初次磁共振成像检查,结果显示右侧横窦存在Borden 2型DAVF,伴有静脉逆流和皮质静脉回流。在左侧罗森塔尔基底静脉(BVR)中也发现了与血流相关的小动脉瘤。两天后,他的运动能力恶化,出现了右侧偏身感觉丧失和严重的丘脑疼痛。失语症不典型,类似经皮质型。重复成像显示BVR的血栓形成动脉瘤扩大,对丘脑囊区有占位效应和水肿。初始的抗水肿措施逆转了神经功能缺损;然而,症状复发,需要紧急进行血管内介入治疗。血管造影显示右侧横窦-乙状窦和窦汇存在广泛的高流量DAVF,主要通过逆行静脉引流至深部静脉和左侧BVR。他接受了分期DAVF栓塞治疗,分流明显闭塞。术后神经功能缺损有所改善,患者在随访6个月时临床无症状。

结论

以反复出现的丘脑症状为主的DAVF罕见。非典型经皮质失语而非流利性失语是丘脑病变的一种不寻常临床表现,可能是由于特定丘脑核受累所致。在存在静脉异常的情况下,优先高流量流入BVR可能会导致静脉壁结构薄弱并促使动脉瘤形成。DAVF栓塞有可能逆转这种不寻常的神经状况。

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