Odontogenic keratocysts are benign intraosseous tumors of odontogenic origin that occur most commonly in the jaw. In particular, they have a predilection for the angle and ascending ramus of the mandible. In contrast, odontogenic keratocysts arising in the maxillary sinus are relatively rare. Two such cases are reported herein. In addition, the English literature that concerns odontogenic keratocysts of the maxillary sinus is reviewed.