頻回の出血を繰り返したと思われるMixed Vascular Malformationの1小児例

A Case of Frequent Intracerebral Hemorrhage from Mixed Vascular Malformation in a Child

I．緒言

　　脳血管奇形（cerebrovascular malformation：CVM）は，組織学的に，arteriovenous malforma-tion（AVM），cavernous angioma（CA），venousangioma（venou malformation：VM）およびin-tracerebral capillar telangiectasia（ICT）の4型に分類されてきた^10,15）．しかし，これらの4型のいずれに分類すべきか困難であったり，同一症例で2つ以上のCVMが隣接して認められたとする報告が散見される^{1,2,4,5,8-14,17-20）}．われわれは，頻回の出血を繰り返したCVMで，組織学的にVMとICTの混在した稀な1例を経験したので報告する．

We reported a rare case of frequent intracerebral hemorrhage from mixed vascular malformation in aninfant. A 1-year and 4-month-old girl was admitted with seizure. She had been diagnosed as having febrileconvulsion at the age of 8 months. Computed tomography (CT) indicated a multicystic mass without perifo-cal edema in the right frontal lobe. The mass was not enhanced by contrast media, but T₁-weighted magne-tic resonance imaging (MRI) revealed a multicystic mass with mixed signal intensity, while T₂-weightedimage demonstrated a heterogenous high signal intensity mass with a marginal low intensity rim. Thesefindings suggested a cavernous angioma causing intracerebral hematomas. Because of this, total resectionwas achieved via a right fronto-temporal craniotomy without neurological deficits. Histologically, numerousdilated venous-type blood vessels interspersed within the normal brain parenchyma were seen. In parts, thevessel walls were abnormally thickened and the lesion coalesced with capillary-like vessels. We diagnosedmixed vascular malformation, mainly composed of venous malformation along with telangiectasia. Thenumber of reports for this so-called mixed vascular malformation have thus been increased. The histopa-thological criteria for classification of vascular malformation should be reinvestigated carefully.