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Neurological Surgery No Shinkei Geka Volume 19, Issue 10 （October 1991）

Neurological Surgery 脳神経外科 19巻10号（1991年10月発行）

Japanese English

症例

脳膿瘍を合併したOsler-Weber-Rendu病の1症例—脳膿瘍発症機序およびその治療法に関する考察 A Case of Osler-Weber-Rendu Disease with Brain Abscess;The mechanism of the formation of brain abscess and its treatment in Osler-Welber-Rendu disease 樋口真秀 ¹ , 大西丘倫 ¹ , 有田憲生 ¹ , 早川徹 ¹ , 池田卓也 ¹ , 最上平太郎 ¹ Masahide HIGUCHI ¹ , Takanori OHNISHI ¹ , Norio ARITA ¹ , Toru HAYAKAWA ¹ , Takuya IKEDA ¹ , Heitaro MOGAMI ¹ ¹大阪大学医学部脳神経外科 ¹Department of Neurosurgery,Osaka University Medical School キーワード： Brain abscess , Osler-Weber-Rendu disease , Pulmonary arteriovenous fistula , Surgical treatment Keyword: Brain abscess , Osler-Weber-Rendu disease , Pulmonary arteriovenous fistula , Surgical treatment pp.969-974

発行日 1991年10月10日 Published Date 1991/10/10

DOI https://doi.org/10.11477/mf.1436900340

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Abstract 文献概要
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I．はじめに

　Osler-Weber-Rendu病（遺伝性出血性毛細血管拡張症，以下O-W—R病と略す）は，皮膚・粘膜の多発性毛細血管拡張（telangiectasia），その部位からの頻回の出血及び家族内発生を主徴とする疾患で，遺伝形式は常染色体優性遺伝とされている^6）．本症には種々の合併疾患が見られるが，その中でも脳膿瘍は治療及びその発生機序において，controversialな点が多い^9,10,13）．今回われわれは肺動静脈瘻，収縮性心嚢炎及び脳膿瘍を合併したO-W—R病の1症例を経験し，さらに剖検を行うことができたので，本症と脳膿瘍との関連性及び脳膿瘍に対する治療方針についての考察を加えて報告する．

Abstract

A 54 year-old man, who had a hereditary hemorrha-gic telangiectasia (Osler-Weber-Rendu disease ; O-W-R) accompanied by pulmonary arteriovenous fistulas (PAVFs) and congestive heart failure, developed sei-zure, right hemiparesis and dysphasia. A brain CT scan revealed a cystic lesion with perifocal edema in left frontoparietal lobe. A contrast enhanced CT scan showed a ring-like enhancement. Dynamic CT scans disclosed that the ring in the cortical side was enhanced more thickly than that in the ventricular side.

Considering the severity of the cardio-pulmonary condition, and the deep location of the abscess, we per-formed an echo-guided aspiration and drainage of the abscess under local anesthesia. No bacteria were de-monstrated in the culture of the contents of the abscess. After the surgery, the right hemiparesis and dysphasia were much improved and a CT scan showed the marked reduction of the abscess. However, aroundeight days after the surgery, the patient showed severe pleural effusion due to progressive heart failure and died on the 11th postoperative clay. Autopsy disclosed a shrunken brain abscess, multiple cerebral infarction, multiple PAVFs and severe constrictive pericarditis which was regarded as the cause of death in the pa-tient. In this report, we presented the therapeutic advantage of echo-guided surgery for the treatment of brain abscess in a high-risk patient. We also discussed the mechanism of the formation of brain abscess in pa-tients of O-W-R disease by reviewing published cases.