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Temporomandibular joint (TMJ) ankylosis is commonly seen in childhood and its etiology is multifactorial. Common causes include congenital malformations, use of forceps during delivery, local or systemic infections or systemic autoimmune diseases (rheumatoid arthritis, psoriasis, and sickle cell anemia), burns, and trauma. TMJ ankylosis leads to dentofacial deformity, restricted mouth opening, malocclusion, difficulty in mastication, malnutrition, dental caries, poor oral hygiene, impaired speech, and cosmetic disability. Neglected childhood bilateral TMJ patients present in adulthood with micrognathia, retrognathia, and severe symptoms of obstructive sleep apnea (OSA). They have narrow airway passages due to pseudoglossia, subatmospheric intrapharyngeal pressure, and hypotonicity of the oropharyngeal muscles which make the mask ventilation difficult. Highlights of the management of the case include the use of the awake fiber-optic-assisted intubation, a gold standard for difficult airway management, and incorporation of Brainlab Kick navigation station 18070 (USA) aiding in the precise surgical dissection and preventing injuries to great vessels and nerves. Extubation strategy with preextubation dose of hydrocortisone and dexmedetomidine helped in the smooth recovery. Early recovery and rehabilitation were further facilitated with the use of nasal continuous positive airway pressure postextubation which helped in reducing symptoms of OSA and airway obstruction.