Orbital compartment syndrome (OCS) is a visual threatening ocular emergency. We report a 50-year-old male with acute presentation of OCS, a rare manifestation of idiopathic orbital inflammatory disease. At presentation, high intraocular pressure was reduced by prompt lateral canthotomy and cantholysis. The disease responded to systemic steroids and treatment resulted in good visual outcome. Detail evaluation and early detection and treatment are mandatory to prevent permanent vision loss.