BACKGROUND Extra-intestinal manifestations of inflammatory bowel disease (IBD) include thromboembolic events that can present as deep vein thrombosis, pulmonary embolism, and cerebral venous sinus thrombosis. Cerebral venous sinus thrombosis is a rare complication of IBD that can be associated with high morbidity and mortality. This report is of a case of cerebral venous sinus thrombosis presenting in a young man during a relapse of ulcerative colitis (UC). CASE REPORT A 27-year-old man presented with seizures and focal neurological deficit during a relapse of chronic UC. He was found to have left cerebral venous sinus thrombosis complicated by left frontotemporal infarction that was treated with anticoagulation therapy. CONCLUSIONS Thromboembolic events are well documented extra-intestinal manifestation of IBD. Cerebral venous sinus thrombosis is a rare but serious complication that can be fatal. The correct diagnosis and timely management require a high degree of suspicion in patients with IBD who present with a new-onset headache, focal neurological symptoms, seizure, or altered mental status.