The odontogenic keratocyst tumour (OKCT) etiology is probably closely related to the remnants of the dental lamina. This lesion is of particular interest because of it propensity to recur and aggressive behaviour. It is distinctive not only because of its potential for local recurrence, but also for its unique histopathologic appearance and association with the nevoid basal cell carcinoma syndrome. Two distinct varieties have been described histologically: orthokeratinized (limited) and parakeratinized (destructive). The most common location is in the lower jaw, in the body and ramus region. Recurrence following the surgical treatment of keratocysts of the jaws may present a major problem to the oral surgeon. Treatment is generally classified as conservative, including simple enucleation, with or without curettage or marsupialization or aggressive, with peripheral ostectomy along with chemical curettage with Carnoy’s solution or resection. However, complete removal of the OKCT can be difficult because of the thin friable epithelial lining, limited surgical access, danger of cortical perforation, and the desire to preserve adjacent vital structures. Recurrence of odontogenic keratocysts has been attributed to several mechanisms as incomplete removal of the original cyst lining, growth of a new OKCT from small satellite cysts or the development of an unrelated OKCT in an adjacent region of the jaws that is interpreted as a recurrence. This paper aims to present a case report in a 39 years-old female patient who underwent to several enucleations of keratocysts. The lesion was migrating upward the mandible ramus achieving the condyle. The definitive treatment was the enucleation of OKCT in addition to condyle resection and rehabilitation with a TMJ prosthesis The patient has a 13 months follow up without recurrence and with functional and stable oclusion.