ABSTRACT The odontogenic keratocyst is a developmental odontogenic cyst. It can occur anywhere in the jaws, but it is commonly seen in the posterior part of the mandible. Lesions have a propensity to grow along the internal aspect of the jaws, and clinically observable expansion of bone occurs late. These lesions have a different mechanism of growth as compared with other cysts, and may show varying radiographic appearances. This paper reports a distinctive case of an odontogenic keratocyst in a 33-year-old female patient. The cyst has an unusual radiographic presentation of two unilocular radiolucencies overlapping each other in the left maxillary premolar-molar region. The study also presents a literature review.