Introduction <p>Cerebral venous thrombosis as the first manifestation of Behçet’s Syndrome (BS) is very rare. We describe a case of BS in which all four major cerebral venous sinuses, including the bilateral sigmoid sinuses, transverse sinuses, and superior sagittal sinuses, exhibited severe cerebral venous thrombosis.</p> Case report <p>An 18-year-old man was brought to our hospital with a three-month-old, wide, throbbing headache in both hemispheres and double, blurred vision while looking to the right for the last week. Magnetic Resonance Imaging (MRI) showed extensive thrombosis involving the bilateral sigmoid sinuses, transverse sinuses, and superior sagittal sinus. Collagen tissue diseases and systemic thrombophilia were ruled out. The patient, who had a history of erythema nodosum and genital and oral aphthae, was evaluated as 3/6 (+) positive by the pathergy test, leading to the diagnosis of BS. Prednisolone 48&#xa0;mg/day and azathioprine 2 × 50&#xa0;mg/day were started, and the prednisolone dosage was lowered to be stopped in three to four months. For nine months, anticoagulant medicine was administered. Partial recanalization was seen on control MRI venography.</p> Conclusions <p>It may be life-saving to consider Behçet syndrome as a differential diagnosis in patients with multiple cerebral venous thrombosis.</p>