一側眼症状で発症した脳動静脈シャントを伴う多発性テント部硬膜動静脈瘻の1治療例—血管内治療と開頭術による2期的治療

Multiple Tentorial Dural Arteriovenous Fistulas with Acquired Pial Arteriovenous Fistula Presented with Unilateral Eye Symptoms Successfully Treated by Transarterial Embolization and Direct Surgery:A Case Report

Ⅰ．はじめに

　多発性硬膜動静脈瘻は稀な疾患であり，両側性の海綿静脈洞病変を除いて，硬膜動静脈瘻（dural arteriovenous fistula：dAVF）全体の約7〜8%と報告されている¹⁾．今回，一側の結膜充血で発症した多発性dAVFsの1例を経験した．皮質静脈逆流（cortical venous reflux：CVR）を伴い，pial AVFを合併した左テント部dAVFに対して経動脈的塞栓術と開頭術で治療し，良好な転帰を得たので，文献的考察を加えて報告する．

　A 58-year-old man presented with slowly progressive chemosis, exophthalmos, and blepharedema of the left eye. Digital subtraction angiography revealed multiple dural arteriovenous fistulas（dAVFs）involving the bilateral tentorium, superior sagittal sinus, and right frontal convexity. In addition, occlusion of the left cavernous sinus and left sigmoid sinus were observed, which could lead to venous hypertension and might be associated with the occurrence of multiple dAVFs. Along with the congestion of the left ophthalmic vein, increased intracranial pressure due to the left tentorial dAVF with cortical venous reflux could have caused the left eye symptoms；therefore, the tentorial lesion was treated first. Although the proximal occlusion of the vein of Labbe, drainer of the left tentorial dAVF, was achieved after embolization using n-butyl cyanoacrylate via the external carotid artery, an arteriovenous shunt between the middle cerebral artery and the distal site of the vein of Labbe was newly visualized after embolization. The newly visualized lesion was treated with further surgical intervention. A pial AVF arising from several peripheral branches of the left middle cerebral artery with drainage into the distal site of the vein of Labbe was confirmed during the surgery, and interruption of the vein of Labbe was performed. The left eye symptoms of the patient significantly improved after the surgery, and postoperative angiogram revealed no residual shunt. Thus, the patient was treated using a combined approach of endovascular and direct surgeries, after which he recovered uneventfully.