Intracranial hypotension is a well recognized disorder of cerebrospinal fluid CSF hypovolemia caused by iatrogenic or spontaneous spinal CSF leakage. The most common manifestation of intracranial hypotension is orthostatic headache, frequently accompanying general fatigue, appetite loss, tinnitus, hearing disturbance, blurred vision, and or limbs dysesthesia. Persistent low intracranial pressure status can lead to several complications, such as cranial nerve palsies, subdural effusion hematoma,

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... cerebellar tonsil descent, superficial siderosis, and brainstem dysfunction, including coma. The occurrence of cerebral venous thrombosis has been reported in some patients with intracranial hypotension, but the mechanism has not been fully elucidated. When these two pathologies occur together, it raises practical problems about the treatment. We present two cases of spontaneous CSF hypovolemia complicated by intracranial hypotension and cerebral venous thrombosis. In one case, extensive venous thrombosis involved many cerebral cortical veins, the superior sagittal sinus, and the right transverse sigmoid sinus, which caused symptomatic diffuse cerebral blood flow disturbance. Treatment with epidural blood patch caused complete resolution of the CSF hypovolemia however, the non orthostatic headache recurred and persisted. Additional anticoagulation was needed for progressive venous thrombosis. In the other case, many cerebral cortical veins, the superior sagittal sinus, and the right transverse sigmoid sinus were involved asymptomatically. Epidural blood patch resolved both the CSF hypovolemia and cerebral venous thrombosis. Cerebral venous thrombosis is a rare but serious complication of CSF hypovolemia. A change in headache pattern has been reported as a possible predictor of the development of cerebral venous thrombosis in patients with intracranial hypotension. We discuss the importance of early detection of cerebral venous thrombosis in patients with CSF hypovolemia along with its management and potential complication.