Introduction: Visual manifestations in pituitary adenoma range from no presenting symptom to visual acuity deterioration to blindness. The presentation of pituitary adenoma is varied. It classically presents with visual field defects, bitemporal hemianopia being the most common Ocular motility disorders in pituitary adenomas are rare presentations. Pituitary adenoma presenting with oculomotor palsies as an initial symptom is very rare, total ophthalmoplegia being the rarest initial presentation. Aim: To report a rare case of pituitary adenoma with cavernous sinus invasion presenting initially with total ophthalmoplegia.