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An early adolescent with bilateral temporomandibular joint (TMJ) ankylosis presented with obstructive sleep apnoea (OSA), facial asymmetry and reduced mouth opening with an interincisal distance of 2 mm. Baseline polysomnography (PSG) revealed a critically elevated apnoea-hypopnoea index (AHI) of 90.3, consistent with extremely severe OSA. Due to high anaesthetic risk and compromised airway, a staged surgical approach was adopted. A planned tracheostomy was performed during the first-stage procedure to secure the airway with subsequent bilateral mandibular distraction osteogenesis leading to significant improvement in OSA symptoms, nutritional status and airway dimensions. Four months later, bilateral TMJ ankylosis release and interpositional gap arthroplasty using dermis fat were performed. Tracheostomy was maintained for approximately 175 days to ensure airway safety during the interval between staged procedures and because of the anticipated difficulty with reintubation for the subsequent surgery. Postoperatively, the unassisted interincisal opening improved to 35 mm. No postoperative device-related or neurological complications were reported. Decannulation was performed after confirmation of adequate airway patency. Repeat PSG performed at 1 year follow-up demonstrated an AHI of 0.6. This report illustrates the effectiveness of a staged approach in managing TMJ ankylosis complicated with severe OSA. In this case, airway management is a point of practical importance for surgical planning due to anticipated difficult intubation and intraoperative risk.