The gluteal region contains 3 distinct myofascial compartments at risk for development of compartment syndrome. Although rare, the most commonly cited etiology is prolonged immobilization or as a complication of intraoperative positioning. Classic symptoms including localized swelling and pain with passive stretch are common presenting symptoms and distal neurological deficits can be seen in approximately half of the patients. Best practice guidelines are based exclusively on a handful of case series and case reports available. Diagnosis relies on the clinical exam and intracompartmental pressure measurements, and may be aided by angiography or other imaging studies. The mainstay of treatment is surgical decompression and delayed primary closure. Immediate complications include infection, acute renal failure, and death; however, as many as 50% of patients treated with surgical decompression may recover with no functional or neurological deficits. We review the history, etiology, diagnosis, and surgical technique used in the treatment of gluteal compartment syndrome.