Chronic diffuse sclerosing osteomyelitis (CDSO) is a disease that presents a rather unusual clinicoradiographic picture creating diagnostic and therapeutic problems. Involvement of the temporomandibular joint is not frequently observed in CDSO. We report a unique case of a 37-year-old male with cyclic episodes of insidious swelling and progressive trismus for the past 4 years. Each episode was followed by spontaneous remission after 2 weeks. Radiographic examination detected unilateral coarse trabeculae with ground-glass appearance of the left hemimandible and resorption of ipsilateral condyle. Computed tomography demonstrated endosteal sclerosis, cortical thickening, and condylar resorption, while magnetic resonance imaging revealed altered marrow intensity with postcontrast enhancement of the surrounding musculature. Bone histopathology was consistent with the diagnosis of osteomyelitis. Palliative antibiotic therapy and regular follow-up were recommended. Of particular interest in the present case is ipsilateral condylar resorption not associated with any dermato-skeletal conditions, which is uncommon in CDSO of the mandible.

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