[A case of cerebral arteriovenous malformation revealed at repeated subcortical hematoma with initially normal angiogram].

An 18-year-old male admitted to our hospital suffered left temporal subcortical hemorrhage. No abnormality was demonstrated on carotid or vertebral angiography at that time. On the day following the onset, left frontotemporal craniotomy was performed and the subcortical hematoma was evacuated. No vascular malformation was found despite careful investigation. On 30th day after the onset, the repeat cerebral angiography was performed but failed to show any vascular abnormalities. After discharge he was in good health, and had had some follow up, CT were normal except for the hematoma cavity. Just two years after the first operation he suffered a second left temporal hemorrhage. Cerebral angiography was repeated and a temporal arteriovenous malformation (AVM) was found with feeding vessels from the M-1 and M-2 portion of the left middle cerebral artery and from the left anterior choroidal artery, and draining veins to vein of Rosenthal and the straight sinus. One month after the second hemorrhage, left frontotemporal craniotomy was performed and complete excision of the AVM was carried out. Only five cases of AVMs in patients with normal angiograms several years before have been reported previously in the literature. But there are no cases in which surgery has been performed. Differently to those cases, in this case it was investigated operatively whether there was a vascular abnormality at the first hemorrhage. We didn't think, however, that the AVM demonstrated at the second hemorrhage had developed spontaneously because there had been a hemorrhage of unknown origin previous to it. It was assumed that a small angiographically occult AVM connected to the hematoma cavity existed at the time of the first hemorrhage but it was too small to be found even during surgical procedure. Such an angiography occult AVM had been growing for two years, and its growth had probably been facilitated by the presence of the hematoma cavity left after the first operation.