Rhabdomyolysis is a condition in which striated muscle tissue breaks down rapidly and releases muscular cell constituents into extracellular fluid and the circulation. Renal symptoms, such as acute renal failure, are major complications of rhabdomyolysis. However, no previous report of rhabdomyolysis associated with orbital complication has been issued. Here, we report the first patient of rhabdomyolysis presenting as orbital apex syndrome. A 66-year-old man presented with right periorbital swelling with erythematous patches and conjunctival chemosis. In addition, swelling, redness, and vesicles were observed in both lower legs. He was found in a drunken state with the right side of his face pressed against a table. Ophthalmic examination showed right eye fixation in all directions and ischemic change of retina. Blood testing showed elevated muscle enzyme associated with muscle destruction. And computed tomography of the orbit showed swelling of right extraocular muscles and crowding of right orbital apex. Under a diagnosis of rhabdomyolysis-associated orbital apex syndrome and central retinal artery occlusion, intravenous steroid and antibiotics therapy with intraocular pressure-lowering topicals were begun. Clinical presentation, treatment course, and follow-up are discussed.