Perioperative pulmonary edema is a rare complication of maxillofacial surgery. However, this potentially fatal complication may arise during any maxillofacial surgery. Negative pressure pulmonary edema can be caused by upper airway obstruction after operation. When this phenomenon arises, if not treated properly, it progresses rapidly causing fatal outcomes. Because orthognathic surgery is performed mostly on healthy and young patients, surgeons and anesthesiologists might neglect the possibility of such complications. Therefore, we must always take into consideration the possibility of negative pressure pulmonary edema. Careful observation of the patient; accurate knowledge; and active, quick, and noninvasive safe measures are required to treat this malady when it does occur. We report a case of negative pressure pulmonary edema after orthognathic surgery and its successful treatment.