Obstructive sleep apnea syndrome (OSAS) is associated with repetitive nocturnal upper airway obstruction leading to daytime sleepiness, cardiovascular derangements, and can be a debilitating, even life-threatening condition. The most favorable treatment for patients with OSAS is multidisciplinary care by a team that represents various dental and medical disciplines. Prescribed therapies might include weight loss, behavior modification, oral appliances, soft tissue surgery, skeletal surgery, or some combination of approaches. Osteogenesis by mandibular distraction has proved effective in children in the treatment of obstructive apnea syndrome associated with congenital malformations. In the adult, the possibility of using distraction osteogenesis in the management of OSAS remains to be defined. We report a case of an adult patient treated for OSAS secondary to temporomandibular joint ankylosis by mandibular distraction followed by interpositional arthroplasty.