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Background: Cerebral venous sinus thrombosis is a rare form of brain stroke caused by thrombosis in venous sinuses of Dura. This study reports on a patient with venous sinus thrombosis and brucellosis who presented with uncontrolled seizure. Case: A 33-year-old woman with past history of controlled complex partial seizure who presented with headache, asthenia, and uncontrolled seizure for one month is described in this study. She was febrile and in brain CT scan hemorrhagic focus in left posterioparietal and temporal lobe was reported. MRI and MRV proved venous sinus thrombosis in left transverse sinus. In laboratory assessment, brucellosis was confirmed as well. The patient treated with anticoagulant, anti-brucellosis, and antiepileptic agents and discharged in good condition with medication orders. Conclusion: Clinical suspicion and accurate evaluation is the most important clue in the diagnosis and treatment of brucellosis and venous sinus thrombosis, especially in uncontrolled seizure in patients who had previously been under control.