首诊于眼科双侧海绵窦区硬脑膜动静脉瘘一例

doi:10.3877/cma.j.issn.2095-2007.2022.04.006

患者女性，68岁。主因"左眼肿胀，视物不清1个月"就诊于河北医科大学第二医院眼科门诊。患者1个月前被柿子砸伤左眼后出现左眼肿胀，视物不清，无头晕头痛，无恶心呕吐。曾于多家医院就诊，诊为"左眼球钝挫伤"、"双眼结膜炎"，治疗1个月未见好转，视力进一步下降。既往高血压5年，血压最高达180/100 mmHg(1 mmHg＝0.133 kPa)。眼部检查，右眼最佳矫正视力0.3，左眼最佳矫正视力0.12；右眼眼压20.5 mmHg，左眼眼压28.0 mmHg；右眼球突出度14 mm，左眼球突出度16 mm；右眼外转活动受限，左眼外转、内转、上转活动均受限；左眼上睑遮盖角膜6 mm；双眼球结膜血管迂曲扩张，左眼球结膜水肿，双眼角膜、前房及晶状体均未见异常。见图1A。右眼瞳孔直径3 mm，对光反射灵敏，左眼瞳孔直径4 mm，对光反射迟钝；眼底检查，双眼视网膜静脉迂曲(左眼重于右眼)，余未见异常。双眼彩色多普勒超声(color doppler flow imaging，CDFI)提示双侧眼上静脉扩张，为动脉型血流频谱。见图1B、图1C及图1D。眼眶磁共振成像提示双侧眼上静脉扩张，左眼下直肌、外直肌、上直肌、上睑提肌肿胀增粗，其周围有渗出性改变。见图1E。头颅磁共振动脉造影和磁共振静脉造影提示海绵窦显影，海绵间窦开放，眼上静脉显影增粗。见图1F。初步诊断为颈动脉海绵窦瘘(carotid－cavernous fistula，CCF)。转入神经内科行全脑数字减影血管造影(digital subtraction angiography，DSA)检查结果显示(1)双侧颌内动脉、脑膜中动脉及咽升动脉等颈外动脉多个分支供血海绵窦区硬脑膜动静脉瘘(cavernous sinus area dural arteriovenous fistula，CSDAVF)，瘘口部位结构复杂；(2)海绵间窦开放，双侧岩下窦与眼上静脉引流；(3)右侧颈内动脉C6段硬脑膜分支供血CSDAVF；(4)左侧颈内动脉C4段海绵窦下外侧干分支供血CSDAVF。见图1G、图1H、图1I。确诊为双侧CSDAVF，于神经内科行硬脑膜动静脉瘘血管栓塞术治疗。术后3个月复查，右眼最佳矫正视力0.6，左眼最佳矫正视力0.3；右眼眼压18.0 mmHg，左眼眼压14.0 mmHg；左眼上睑遮盖角膜2 mm，左眼球结膜水肿消退，双眼球结膜血管无迂曲扩张。见图1J。双眼瞳孔直径3 mm，相对性传入性瞳孔障碍(－)，眼球运动较前好转；双眼视网膜静脉无迂曲扩张；双眼CDFI复查结果显示双侧眼上静脉均未见扩张。见图1K。DSA显示双侧CSDAVF栓塞完全，无残余瘘显影。

图1 海绵窦区硬脑膜动静脉瘘患者治疗前后的临床表现及辅助检查结果　图A示左眼上睑下垂，双眼结膜血管迂曲扩张，左眼结膜水肿；图B示超声检查发现双侧球后壁弥漫增厚，球后管状低回声，提示双侧眼上静脉扩张；图C示双眼彩色多普勒超声示扩张的眼上静脉血流信号丰富；图D示双眼彩色多普勒超声示扩张的眼上静脉为动脉型血流频谱；图E示磁共振成像见眼上静脉显影、海绵窦显影及海绵间窦开放；图F示磁共振动脉成像见动脉显影基础上，双侧异常增粗显影的眼上静脉；磁共振静脉成像静脉窦期见异常显影的眼上静脉；图G示右侧颈外动脉侧位全脑数字减影血管造影见参与供血动脉有脑膜中动脉(红色箭头)、脑膜副动脉(黄色箭头)及颌内动脉(绿色箭头)。引流静脉为岩下窦向右颈内静脉引流(蓝色箭头)；图H示左侧颈内动脉侧位全脑数字减影血管造影见参与供血动脉有脑膜垂体干(红色箭头)和经岩下窦向左侧颈内静脉引流(粉色箭头)；图I示左侧颈外动脉侧位全脑数字减影血管造影见参与供血的动脉有脑膜中动脉(红色箭头)、脑膜副动脉(黄色箭头)及颌内动脉(绿色箭头)。经岩下窦向右颈内静脉引流(蓝色箭头)和眼上静脉引流(紫色箭头)；图J示术后3个月双眼结膜充血消失，左眼上睑下垂好转；图K示术后双眼超声检查见球后管状低回声消失，提示双侧眼上静脉扩张减轻

表1 海绵窦区硬脑膜动静脉瘘和颈动脉海绵窦瘘鉴别诊断

项目	海绵窦区硬脑膜动静脉瘘	颈动脉海绵窦瘘
发病年龄	中老年	年轻
病因	不明，颅内静脉窦血栓、静脉窦高压、炎症、外伤、内分泌因素等继发	多于外伤有关、颅底骨折
性别	女性多见	男性多见
病程	缓慢发展	发展迅速
瘘口解剖	绝大多数属于低流高阻痿，痿口小，数量多，供血动脉多	一般为高流量直接瘘
颅内血液动力学改变	少见	多见
搏动性眼球突出	少见	多见
颅内血管杂音	少见	多见
结膜充血水肿的程度	轻	重

表1 海绵窦区硬脑膜动静脉瘘和颈动脉海绵窦瘘鉴别诊断

项目	海绵窦区硬脑膜动静脉瘘	颈动脉海绵窦瘘
发病年龄	中老年	年轻
病因	不明，颅内静脉窦血栓、静脉窦高压、炎症、外伤、内分泌因素等继发	多于外伤有关、颅底骨折
性别	女性多见	男性多见
病程	缓慢发展	发展迅速
瘘口解剖	绝大多数属于低流高阻痿，痿口小，数量多，供血动脉多	一般为高流量直接瘘
颅内血液动力学改变	少见	多见
搏动性眼球突出	少见	多见
颅内血管杂音	少见	多见
结膜充血水肿的程度	轻	重

表2 海绵窦区硬脑膜动静脉瘘的分类

Borden分级	Cognard分级
Ⅰ级，脑通过膜静脉或静脉窦引流	Ⅰ级，硬脑膜向静脉窦正向引流
	Ⅱ级，不充分的静脉窦引流
	Ⅱa级，硬脑膜向静脉窦逆向引流
Ⅱ级，通过静脉窦、皮层静脉和软膜静脉引流	Ⅱb级，硬脑膜向静脉窦正向引流+皮层静脉和软膜静脉引流
	Ⅱa+b级，硬脑膜向静脉窦逆向引流+皮层静脉和软膜静脉引流
Ⅲ级，通过皮层静脉和软膜静脉引流	Ⅲ级，仅皮层静脉和软膜静脉引流，无静脉扩张
	Ⅳ级，仅皮层静脉和软膜静脉引流同时伴有静脉扩张
	Ⅴ级，脊髓髓周静脉引流

表2 海绵窦区硬脑膜动静脉瘘的分类

Borden分级	Cognard分级
Ⅰ级，脑通过膜静脉或静脉窦引流	Ⅰ级，硬脑膜向静脉窦正向引流
	Ⅱ级，不充分的静脉窦引流
	Ⅱa级，硬脑膜向静脉窦逆向引流
Ⅱ级，通过静脉窦、皮层静脉和软膜静脉引流	Ⅱb级，硬脑膜向静脉窦正向引流+皮层静脉和软膜静脉引流
	Ⅱa+b级，硬脑膜向静脉窦逆向引流+皮层静脉和软膜静脉引流
Ⅲ级，通过皮层静脉和软膜静脉引流	Ⅲ级，仅皮层静脉和软膜静脉引流，无静脉扩张
	Ⅳ级，仅皮层静脉和软膜静脉引流同时伴有静脉扩张
	Ⅴ级，脊髓髓周静脉引流

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One case of bilateral cavernous sinus dural arteriovenous fistulaat initial diagnosis in ophthalmology

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One case of bilateral cavernous sinus dural arteriovenous fistulaat initial diagnosis in ophthalmology