: Several cases of hypothyroidism have been reported to develop idiopathic intracranial hypertension not directly precisely linked with cerebral venous sinus thrombosis (CVT). A 31-year-old Chinese woman presented with bilateral blurred vision and paroxysmal amaurosis for about 6 months without headache. Neurological examination revealed normal expect for the sixth cranial nerve palsy and bilateral papilledema. Laboratory tests showed pronounced hypothyroidism and greatly increased serum triglyceride. Cerebral spinal fluid showed the increased opening and closing pressure. Digital subtraction angiography (DSA) disclosed a filling defect in the adjunction of bilateral transverse sinuses and sigmoid sinuses. Her symptoms gradually improved with levothyroxine, mannitol and anticoagulants treatment. In presenting the rare case of lateral sinus thrombosis associated with primary hypothyroidism, we wish to alert physicians that patients presenting with papilledema and hypothyroidism may require investigations of DSA for CVT, even in the absence of headache.