Primary Tuberculous osteomyelitis involving the mandible represents less than 2% of skeletal locations. In this paper, we report a case of mandibular tuberculosis detected after histopathological analysis of the surgically resected specimen during surgical management of a suspected case of ameloblastoma. A 14 -year-old male patient presented to us with history of right sided chin swelling. The clinical examination revealed a swelling, involving right body and parasymphysis of mandible, measuring approximately 6cm in length and 2cm in width, extending from right lateral incisor till the first molar. Radiological scans revealed a large multiloculated osteolytic expansive lesion measuring 52 x 20 x18 mm. Excision of the lesion was performed and reconstruction was done with iliac bone grafting. The histopathological findings revealed a granulomatous lesion, suggestive of tuberculous osteomyelitis. The patient was successfully treated with standard multidrug therapy. One year after completion of therapy, there were no signs of recurrence. Primary mandibular tuberculosis is an extremely rare entity. Its clinical presentation is not specific. Radiologically, tuberculosis has no characteristic appearance. The positive diagnosis is based on histology. Primary mandibular tuberculosis is rare and should be kept amongst differential diagnoses in susceptible population and in endemic areas.