Keratocystic odontogenic tumor (KCOT), formerly known as the odontogenic keratocyst (OKC) is a benign intraosseous lesion, characterized by invasive growth and high rate of recurrences. Although it is not frequently seen, KCOT may be associated with unerupted teeth and without relevant histopathological examinations, might be mistaken with dentigerous cyst. A 26 year-old male patient referred to our department with complaints of two months persisting painless swelling on the left side of mandible. Clinical and radiographic examinations revealed an impaction of the horizontally positioned left lower second premolar surrounded by a well-circumscribed radiolucent mass, located between the left canine and the second molar, suspected to be a cyst. The aim of this report is to present a therapeutical approach to keratocystic odontogenic tumor associated with impacted tooth and prosthetic rehabilitation with dental implants after an initial treatment. The patient is under follow up for 31 months since implant placement was done, without any complaints or complications