Neurological symptoms are rare manifestations of Behcet's disease. In this paper, we report cerebral venous thrombosis as the initial presentation of Behcet's disease in a 40 yearold man. Our patient presented with a six-week history of headache, progressive loss of vision in the right eye and recurrent oral ulcers. Physical examination revealed oral ulcers and posterior synechia in the right eye with loss of light perception. Fundoscopic examination showed evidence of retinal vasculitis. Magnetic resonance imaging (MRI) and magnetic resonance venography (MRV) of the brain showed superior sagittal, left transverse, and left sigmoid sinus venous thrombosis. A diagnosis of Behcet's disease was made based on clinical criteria. Treatment with methylpredinsolone, cyclophosphamide, and azathioprine as well as anticoagulation was done with significant clinical improvement.

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