Leptospirosis is an acute infectious disease caused by a spirochete of the leptospira gender and it is characterized by severe vasculitis. It is not common for leptospirosis to present as a primary neurological disease. On the other hand, no study has been reported on the progression of cerebral venous thrombosis (CVT) in patients with leptospirosis so far. This is the first case reported which shows CVT as a complication after the leptospirosis infection. An acutely developed leptospirosis and post-infectious CVT in a 21-year-old soldier is described here.