A 21-year-old man had been diagnosed as having thrombosis of the dural sinus secondary to Behçet's disease. Despite treatment with acetozolamide, azathioprine and methylprednisolone over the ensuing 6 months, he had little improvement in his symptoms of intracranial hypertension and required frequent lumbar CFS drainage. The patient then underwent lumboperitoneal shunt operation, which resulted in rapid resolution of papilledema and gradual improvement in the visual field. In conclusion, we suggest that in BD patients with dural sinus thrombosis, in whom the intracranial hypertension developed but failed to normalize after treatment with immunosuppressive agents, lumboperitoneal shunting should be considered as an effective therapeutic tool.