
Introduction: Primary tuberculous osteomyelitis of mandible is an extremely rare condition, particularly in comparison to pyogenic infections and neoplastic diseases involving the mandible. Osteoarticular tuberculosis accounts for 1–2% of all the types of bone tuberculosis. Bone tuberculosis forms about 10% of extrapulmonary tuberculosis, of which 50% occur in the spine. Tuberculosis of the flat bones of the skull is uncommon and that of the mandible is especially rare as it contains less cancellous bone. Case report: A 9 year old female patient presented with complaint of gradually increasing swelling and persisting pus discharge from sinus on left lateral border of mandible and submandibular region since 2 months with accompanying low grade fever. Extraoral examination revealed a unilateral diffuse swelling over the left body of the mandible. The overlying skin was erythematous and showed the presence of a fistula with a serosanguinous discharge. Routine laboratory investigations were grossly unremarkable with raised total leucocyte count. There was no evidence of regional lymphadenopathy. Radiographic examination revealed carious deciduous molars with periapical abscess, CT mandible revealed marked periosteal thickening with change in trabecular pattern followed by chest examination and radiograph. Curettings from the lesion sent for histopathological examination showed collection of epitheloid cells alongwith Langhan type of giant cell and multiple granulomas. Tentative diagnosis of primary tuberculous osteomyelitis of the mandible was made and patient had been put on antitubercular drug therapy. Due to its rarity and clinical interest one should always have a suspicion of mycobacterial infection in the back of mind when dealing with chronic maxillofacial infections not responding to the usual antibiotic course and local debridement.