A case of a 43-years-old patient with a 9-year history of Behçet's disease is reported. The diagnosis was based on the past of bilateral hypopion iritis, oral aphthous ulceration and venous thrombosis. A right lower limb monoparesia occurred. CT scan and angiography showed a voluminous intracerebral angiodysplasia and an aneurysm of the left anterior communicating artery. Careful angiographic examination of visceral and peripheral arteries showed bilateral thrombosis of subclavian arteries, thrombosis of superior and inferior mesenteric arteries and an aneurysm of the coeliac trunk. Large arterial involvement is an unusual complication of Behçet's disease. A through review of the literature showed only 2 reported cases of intracranial arterial aneurysms. This case report was the first case of Behçet's disease with an intracerebral angiodysplasia.