Odontogenic keratocyst remains an enigma for clinicians and researchers, although gains in knowledge in recent years have improved the understanding of this interesting lesion. The diagnostic problems are mainly related to the relative lack of specific clinical and radiographic features that unequivocally point to a proper diagnosis. Of particular interest to clinicians is the biologic behaviour of keratocysts which includes high rates of recurrence and potential existence as a benign odontogenic neoplasm, keratocystic odontogenic tumour. Various surgical modalities have evolved in an attempt to reduce the recurrence rate, including curettage, peripheral ostectomy, removal of overlying mucosa in cases of cortical perforation and osseous resection in the form of marginal or segmental osteotomies. The present case report describes the conservative surgical management of a large keratocystic odontogenic tumour in a young patient with no evidence of recurrence at two years follow-up.