Introduction: We reported an infant with temporomandibular joint (TMJ) osteoarthritis who required long-term tracheostomy. Case findings: At 1 year of age, he received urgent tracheostomy because of apnea due to pneumonia. The tracheal cannula could not be removed thereafter due to trismus. Computed tomography (CT) images demonstrate irregular bone formation. However, we could not diagnose osteoarthritis of temporomandibular joint because TMJ adhesion was not clear on CT images. Interventions: Arthroplasty was performed under general anesthesia, but there was no significant improvement. A 3-D plastic model based on intraoperative findings showed that atrophic change of the muscles also disturbed movement of the mandible. Therefore, an additional procedure called "pan-muscle-release surgery" was performed, resulting in further improvement of mandible movement. After confirming that mouth opening was stable, the tracheal cannula was removed.
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