Temporomandibular joint ankylosis in children commonly lead to difficulty in feeding, poor oral hygiene, retrognathic mandible and obstructive sleep apnea. Surgical release of the ankylosis has always been the standard treatment. The authors report a 12 year old boy with unilateral temporomandibular joint ankylosis and obstructive sleep apnea underwent surgical release of the ankylosis with successful gain in mouth opening. However, he continued to suffer from obstructive sleep apnea as confirmed by post-operative polysomnography. Orthognathic surgery for mandibular advancement is not favorable due to his young age and mandibular distraction osteogenesis was not a choice. A mandibular advancement device similar to orthodontic myofunctional appliance was the preferred choice in the post-operative period while waiting for definitive retrognathia surgical treatment after skeletal maturity. Surgical release of temporomandibular joint ankylosis corrects the oral problem but does not adequately address the narrow pharyngeal airway space. Assessment of pharyngeal airway with a high suspicion of obstructive sleep apnea is mandatory in the management of TMJ ankylosis.
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