Adenomatoid Odontogenic Tumour is a relatively uncommon benign odontogenic tumor which affects young individuals with a female predominance, mainly in the second decades of life. This lesion is most commonly affected in the anterior maxilla and usually associated with impacted tooth. In this paper, we present a case of extra-follicular AOT associated with dentigerous cyst affecting the mandibular canine-premolar region in a 25 year old female patient without any impacted tooth – a very rare situation.