A 55-year-old man presented with a rare case of multiple isolated sinus dural arteriovenous fistulas (AVFs) associated with antithrombin (AT) III deficiency manifesting as sudden onset of headache and gait disturbance. Increased arterial shunting flow had caused intraventricular hemorrhage after incomplete repeated transarterial embolization procedures for dural AVFs. Multiple isolated sinus dural AVFs were located in the anterior superior sagittal sinus (SSS) and transverse sinus, which were completely embolized by direct packing of the isolated sinuses via the SSS. The development of dural AVF is complicated and associated with a number of factors, such as congenital abnormality, head trauma, craniotomy, radiation, hematological abnormality, and sinus thrombosis. Hematological abnormality is a risk factor of sinus thrombosis. In the present case, the multiple isolated sinus dural AVFs might have resulted from the aggravation of multiple dural AVFs and the coagulative tendency due to AT III deficiency. Direct sinus packing should be considered if transvenous catheterization is difficult or fails.