特発性海綿洞動静脈瘻に施行した脳血管撮影術中Diffuse Cerebral Telangiectasiaから致命的脳出血をきたした1例

Fatal Ruputure of Diffuse Cerebral Telangiectasia in a Woman Associated with a Spontaneous Carotid Cavernous Fistula During Cerebral Angiography

I．はじめに

　海綿洞動静脈瘤の約1／4は特発性といわれ^6），そのなかのあるものは内頸動脈瘤破裂によるもの^3），あるいは粥状硬化性病変が基盤となつているもの^3）などが知られている。この種の疾患に対する術前準備としての脳血管撮影術施行の必然性は，こんにちでは明白なところであるが，このさいの合併症は重篤なことが多く，Hamby^4）によれば，かれの手術合併症のそれに匹敵したという。しかしながら，この合併症のpathogenesisについての報告は少ないように思われる。

　著者らは，左特発性海綿洞動静脈瘻の患者に施行した第1回目の頸動脈撮影で，左前頭側頭部にdifuse cere—bral telanglectaslaが認められ，数日後に施行した第2回目の頸動脈撮影術中，致命的脳内出血をきたした症例の，剖見的，病理組織学的所見について報告する。

The pathogenesis of complications of angiography performed as preoperative management for a carotid-cavernous fistula has not frequently been reported. The complications were emphasized to have been as severe as were those of surgery in Hamby's series. The present authors' intention is to report post-mortem analysis of such a case.

CASE REPORT : Seven weeks prior to admission, a woman aged 28 developed a spontaneous carotid-cavernous fistula on the left. On the third day after the admission left carotid angiography was carried out, showing diffuse cerebral telangiectasia as well as a carotid-cavernous communication through an aneurysm. One week later, serial angiography was done so as to get a detailed knowledge of vascular system and an abnormal vascular nest. After the angiography, the patient was found to be comatous, and finally expired in spite of intensive treatment 48 hours after the onset.

Autopsy disclosed marked edema and a large haemorrhage within the left fronto-temporal lobes as well as an aneurysm of the internal carotid artery within the cavernous portion. Microscopic examina-tion of conducting arteries revealed internal sclerosis, much waving or duplication of the internal elastica, and edematous swelling, fibrosis of the media and of the adventitia. By and large, pial arterioles, per-forating arteries as well as capillaries were proved to be not only dilated but also bled.

It might be postulated that haemorrhage was caused by stream strain due to a collision between normal circulation and forced back flowing through the fistula.