A 32-year-old man presented with a rare case of spontaneous intracranial hypotension (SIH) associated with dural sinus thrombosis (DST) manifesting as severe postural headache which was relieved by lying down. Initial cerebrospinal fluid pressure was low. He was treated with hydration and rest under a diagnosis of SIH. Magnetic resonance (MR) imaging after 1 month showed signs of both DST and SIH. However, the patient did not demonstrate any of the classical symptoms associated with DST. The patient underwent an epidural blood patch procedure. His headache was relieved and MR imaging showed improvement of both SIH and DST. This case suggests that epidural blood patch may be effective in some cases of SIH associated with DST.