Odontogenic keratocyst (OKC), also known as keratocystic odontogenic tumor, is a distinct clinicopathologic lesion that can be clearly identified by histologic examination. Clinically, OKC is characterized by a high recurrence rate. This report describes a rare case of OKC with mural calcification in the maxilla of a 47-year-old male patient. Orthopantomography and Cone Beam Computed Tomography showed full opacification with calcification and lateral wall resorption in the left maxillary sinus, destroying the sinus floor. Hard tissue deposits have rarely been reported in the connective tissue walls of OKC. The importance of calcified material formation to the biological behavior of OKCs is unclear. Although its prognostic value has not been studied, the presence of calcification materials does not appear to increase the risk of recurrence. Study of a number of samples would be needed to determine the nature of the correlation between the presence of calcified materials and recurrence.