The aim of this study was to evaluate the efficacy of the Odontogenic Keratocyst treatment through enucleation and cryotherapy with liquid nitrogen in a lesion that had previously been biopsied and also to compare the microscopic characteristic of the enucleated lesion to the biopsy microscopy. Enucleation was performed in a 27-year-old female patient with previously diagnosis by initial biopsy. The lesion extended from left mandibular body region to the ramus ipsilateral. The use of this technique proved to be an effective treatment. It is believed that the initial biopsies allowed a decrease in the size of the lesion and increase the thickness of the fibrous capsule, which prevent the rupture of the keratocyst capsule.