Orbital compartment syndrome is a rare ophthalmic emergency characterized by increased intraorbital pressure and hypoperfusion of critical neural structures, most of which were caused by trauma, and can also be caused by periorbital surgery, local injections, other preexisting medical conditions and so on. It requires rapid identification and immediate treatment for the preservation of vision./r/nA 61-year-old female with left frontal lobe-parafalcine meningioma underwent a craniotomy with a bicoronal incision. Postoperatively, the patient presented absence of pupillary reaction in both eyes, and complained loss of vision after recovery from anesthesia. Bilateral orbital compartment syndrome was considered 18 hours postoperatively since the marked bilateral proptosis with eyelid edema and conjunctival chemosis. The patient was treated with methylprednisolone, mannitol, hyperbric oxygenation, and neurotrophic agents as recommended by the ophthalmologist. There was no improvement in visual acuity at discharge or at 3-month follow-up postoperatively./r/nThis is a rare case of bilateral irreversible blindness caused by orbital compartment syndrome after a craniotomy with coronal incision. Neurosurgeons need to improve the awareness of this complication for adequate prevention, such as direct ocular pressure from skin flaps, congestion from head positioning, and adequate intraoperative eye protection, and also earlier recognition and management.