<h3>Clinical Presentation</h3> We present a case of a 20-year-old male who presented with facial asymmetry, severe trismus since early childhood, generalized physical weakness, and other skeletal deformities, including pigeon chest and kyphoscoliosis. No clear history of trauma was elicited. However, early childhood trauma was considered a possibility. <h3>Differential Diagnosis</h3> Severe trismus can be caused by various etiologies, including trauma, temporomandibular joint (TMJ) disorders, benign and malignant neoplasia, infections, and iatrogenic causes. However, with the history and clinical presentation, a TMJ condition, including bony and fibrous ankyloses, was considered. <h3>Diagnosis and Management</h3> Multidetector computed tomography (MDCT) was performed. Radiographic features were consistent with stage IV bony ankyloses of the left TMJ, according to the classification system of Sawhney et al. A 3-dimensional (3-D) printed medical model of the skull and the mandible was used for surgical simulation. Gap arthroplasty with interpositional temporalis muscle flap was performed, followed by vigorous physiotherapy. After surgery, mouth opening increased significantly from 0 to 35 mm. One year clinical follow-up was done. <h3>Discussion</h3> TMJ ankylosis is a pathologic condition in which the mandible is fused to the glenoid fossa by bony or fibrotic tissues, interfering with mastication, speech, oral hygiene, and nutrition. Multiple factors, including trauma, arthritis, infection, congenital deformities, or iatrogenic causes, can result in ankylosis, but trauma remains the most common etiology. In growing individuals, it can result in very severe craniomaxillofacial deformities and can also affect the airway. This case report discusses the clinical and radiographic features affecting the craniomaxillofacial structures associated with TMJ ankylosis and the applicability of 3-D printing in treatment planning. Because TMJ ankylosis can affect multiple aspects of the patient's life, an interdisciplinary management approach is required.
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