ABSTRACT
Odontogenic Keratocyst (OKC) is an odontogenic cyst of developmental origin arising from remnants of the dental lamina. Malignant or benign transformations though rare have been noticed from their epithelium. Ameloblastomatous transformation from an OKC is extremely rare with such lesions being referred to as combined/"hybrid" odontogenic lesions. In this article, we present an intriguing case of a 60-year-old male who was operated on for OKC of the anterior mandible 3 years before, who came back with a complaint of swelling over the same site. Incisional biopsy revealed the acanthomatous type of ameloblastoma for which segmental resection with immediate reconstruction using recon plate was done. Excision biopsy revealed a plexiform variant as well, thus exhibiting a hybrid pattern. Surgeons should be aware of this hybrid presentation of ameloblastoma arising from the epithelium of OKC as the acanthomatous subtype is known for its notorious genetic behavior leading to recurrence and aggressive nature of this tumor.
