A 27-year-old man suffered from acute blindness with pain in the left eye after 4 hours of prone-positioned spinal surgery under general anesthesia. Rapid progression of proptosis, a swollen eyelid and severe conjunctival chemosis were noted, while visual acuity in the left eye indicated light perception only. Orbital compartment syndrome (OCS) was suspected, and an urgent orbital computed tomography (OCT) scan demonstrated globe tenting and swelling of all extraocular rectus muscles. Fundus examination revealed optic disc edema and a cherry-red spot in the macula. Over the 6-month follow-up period, the proptosis and swollen extraocular muscles completely subsided, but the left eye continued to exhibit an absence of light perception.