Objective: This study aims to analyze the clinical, laboratory findings, treatments and prognosis of Behçet’s disease (BD)-associated cerebral venous sinus thrombosis (CVST) and to compare the clinical features of BD patients with and without CVST. Methods: In this single-center retrospective study, we reported a series of 24 consecutive CVST patients (20 males and 4 females; mean age 34±12 years) were diagnosed with BD according to international study group criteria. The control group included 36 (24 males and 12 females; mean age 32±8 years) consecutive patients with BD without CVST from the same center. Results: Headache (n=20, 83.3%) was the most common complaint at admission in patients with BD-associated CVST. In comparison between BD cases with and without CVST, extracranial vascular involvement was more frequent in the BD-associated CVST patients (p=0.03). Twentythree (95.8%) patients received corticosteroid (CS) treatment. A total of 8 BD-associated CVST patients received anti-tumor necrosis factoralpha (anti TNF-α) treatment. There was no significant difference in the rate of CS use before and after anti-tumor necrosis factor-α (TNF-α) treatment (p=0.345) but CS dose was significantly reduced after treatment (p=0.018). When patients with BD- associated CVST who receive or did not receive anti-TNF-α treatment compared, on cranial imaging, thrombosis was significantly less in patients received anti TNF-α treatment than who did not (p=0.02). Conclusion: When the CVST was detected in young men patients with headache, BD should come to mind and patients should also be evaluated in this respect. As the risk of extracranial vascular involvement increases in such patients, they should be systematically evaluated in terms of vascular involvement. Although CS treatment could not be stopped completely with anti TNF-α drugs, it could be reduced. Significant radiological improvement was observed with antiTNF-α treatment.