A 50-year-old man was treated with stereotactic gamma knife radiosurgery for an incidentally detected small callosal arteriovenous malformation (AVM) with stenosis of the main draining pathway. He suffered two episodes of intraventricular hemorrhage at 4 and 14 weeks after the radiosurgery. Radiological studies demonstrated that the anterior portion of the draining system including a varix, which had been irradiated, was thrombosed before the obliteration of arteriovenous shunts. Stereotactic radiosurgery is an accepted treatment for selected small and medium AVMs, but this procedure may increase the risk of bleeding. Early occlusion of a part of the draining pathway after stereotactic radiosurgery might have induced AVM rupture. An impaired venous outlet, either consisting of one draining vein or with stenosis, present before treatment may be develop thrombosis in response to high-dose irradiation.