Tehran University of Medical Sciences Case Reports in Clinical Practice 2538-2683 10 1 2025 09 08 1 6 Islam Nasiri Oral and Maxillofacial Department, Tabriz University of Medical Sciences, Tabriz, Iran. Shima Azizi Oral and Maxillofacial Department, Tabriz University of Medical Sciences, Tabriz, Iran. Hosein Eslami Oral and Maxillofacial Department, Tabriz University of Medical Sciences, Tabriz, Iran. Houman Sojoudi Oral and Maxillofacial Department, Tabriz University of Medical Sciences, Tabriz, Iran. 2025 04 27 2025 06 30 Temporomandibular joint (TMJ) ankylosis is a debilitating condition characterized by the fusion of the  mandibular condyle to the glenoid fossa, leading to restricted mandibular motion. We report the case of a 26-year-old female with left-sided TMJ ankylosis, presenting with limitation in mouth opening and degenerative changes. Preoperative imaging and planning facilitated the fabrication of patient-specific cutting guides and custom alloplastic components. Surgical intervention involved careful osteotomies, resection of the ankylotic mass, and placement of custom joint components into the surgically prepared fossa. Postoperatively, the  patient underwent intermaxillary fixation for 2 months, and was then referred to a physiotherapist, ultimately achieving a mouth opening of 30 mm. Follow-up at 1 and 3 months showed stable improvement, with no signs of reankylosis. Customized TMJ reconstruction, combined with an autogenous fat graft, offers a viable solution for restoring mandibular mobility and improving quality of life in patients with TMJ ankylosis. https://crcp.tums.ac.ir/index.php/crcp/article/view/1042 https://crcp.tums.ac.ir/index.php/crcp/article/download/1042/703