Objective To explore the key points of diagnosis and treatment of Behcet's disease with venous sinus thrombosis.Methods The diagnosis and treatment of a 25 year-old female patient with recurrent headaches was analyzed and discussed.Results Her headache was due to Behcet's disease with venous sinus thrombosis and her symptoms were gradually relieved.Conclusion Physician should be asked about the history in patients with persistent headache and the correct diagnosis can be made by MRI.