Odontogenic lesions are diverse pathologies of jaw representing a wide variety of lesions which develop from remnants of tissue associated with odontogenesis. It includes various cysts which are commonly encountered in dental practise and tumours which are relatively uncomon accounting for less than 1%. These can develop at any age and any site of the jaws. They may remain occult or can be aggressive. We report a unique case of extensive multilocular lesion of the mandible with cortical bone expansion and root resorption in a 40 year old female. Although the closest differential diagnosis was ameloblastoma, the histopathological examination revealed a cystic lesion with diverse features which was finally concluded as Keratocystic odontogenic tumour. The present case is unusual in terms of its site of occurrence, presentation and histopathology emphasizing the importance of incisional biopsies from multiple sites in case of extensively large lesions and meticulous histopathological examination of the entire lesion for establishing a final diagnosis.
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