A 32-year old female patient with a chief complaint of headache during the last 1 year and bilateral visual loss in the last 3 months was evaluated in our clinic. The visual acuity was light perception OU and there was optic atrophy OU. The cranial MR revealed thrombosis of right transverse sinus and further workup revealed hyperhomocystinemia, as the underlying cause. Despite vigorous medical and surgical treatment there was no improvement in the visual acuity of the patient.