Introduction. Immune thrombocytopenic purpura is an autoimmune hematological disorder characterized by low platelet level due to its destruction through autoimmune antibodies. Cerebral venous thrombosis is a serious condition defined by a thrombosis in the cerebral venous sinuses that occurs mostly in the presence of a hypercoagulable state. Hemorrhage and thrombosis are processes with a paradoxical etiology; thus, the association between these two conditions seems to be extremely rare. Case Presentation. We herein report a case of a 19-year-old female with a chief compliant of generalized tonic-clonic episode, severe headache, and blurred vision. Physical examination was significant for a bilateral Babinski’s sign and severe bilateral papilledema. Laboratory workup, computed tomography, and magnetic resonance imaging were normal except for severe thrombocytopenia. Magnetic resonance venography was diagnostic for cerebral venous thrombosis. Her past medical history was significant for immune thrombocytopenic purpura that was treated with prednisolone 40 mg per day which posed a therapeutic challenge. High-dose prednisolone and platelet transfusion were initiated; enoxaparin was administrated and switched to warfarin after stabilization of platelet count. The patient was neurologically intact after 14 days of appropriate treatment and was on follow-up. Many hypotheses were suggested to explain the unexpected thrombotic events in a patient with immune thrombocytopenic purpura which were related to the disease etiology or treatment, taking into account common risk factors (such as age, obesity, smoking, hypertension, diabetes mellitus, dyslipidemia, splenectomy, and oral contraceptive agents). Conclusion. The association between immune thrombocytopenic purpura (which is a major risk factor for bleeding) and cerebral venous thrombosis ( which is caused by a thromboembolic event )has carried a major challenge to the management plan. We believe that immune thrombocytopenic purpura and its treatment methods should be taken into consideration as risk factors for thromboembolic phenomenon.