Obesity hypoventilation syndrome (OHS) is defined by the triad of obesity, daytime hypoventilation, and sleep-disordered breathing without an alternative neuromuscular, mechanical, or metabolic cause of hypoventilation. It is a disease entity distinct from simple obesity and obstructive sleep apnea. OHS is often undiagnosed but its prevalence is estimated to be 10-20% in obese patients with obstructive sleep apnea and 0.15-0.3% in the general adult population. Compared with eucapnic obese patients, those with OHS present with severe upper airway obstruction, restrictive chest physiology, blunted central respiratory drive, pulmonary hypertension, and increased mortality. The mainstay of therapy is noninvasive positive airway pressure. Currently, information regarding OHS is extremely limited in the anesthesiology literature. This review will examine the epidemiology, pathophysiology, clinical characteristics, screening, and treatment of OHS. Perioperative management of OHS will be discussed last.