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Aim Synovial Chondromatosis (SC) is a rare, benign non neoplastic arthopathy characterized by the metaplastic development of cartilaginous nodules within the synovial membrane. In only 3% of all cases does it affect the temporomandibular joint (TMJ) and cases that arise from the lower compartment are rarely found in literature. The aim of this paper is to report a new case of SC of the inferior TMJ compartment with the description of the clinical, therapeutic and histopathological findings. Case report This article presents a 68-year-old woman with preauricular swelling on the right side, pain, crepitus and limited joint motion. This patient was evaluated by preoperative clinical manifestation, CT scan and MR images. Both showed multiple, calcified loose bodies in the inferior compartment. Based on these images as well as the patient's signs and symptoms, a surgical intervention was performed. A good functional recovery with no signs of recurrence at 36 months of follow up was obtained. Conclusion Among cases of synovial chondromatosis in literature, only twelve originating in the lower compartment have been reported, this one included. In all the cases treated for SC in the lower compartment, both in literature and in our case report, surgical treatment led to healing.