Ameloblastoma is a benign odontogenic tumour of epithelial origin and comprises 1% of maxillomandibular tumors or cysts. The incidence of pathological changes such as ameloblastoma from the follicle of impacted third molar was reported to have low incidence. However, there are many reports that asymptomatic third molars are related with various pathological conditions. A case of ameloblastoma secondary to third molar extraction and subsequent sagittal split ramus osteotomy (SSRO) had not been reported. At the right ramus area, radiolucent lesion had been noted at 6 years after the surgical extraction of the third molar followed by SSRO for the mandibular prognathism. The lesion was proved to be the basal cell type ameloblastoma. There had been no significant bony lesion before or 1 year after the SSRO. The tumour was successfully removed and there was no evident recurrence at 4 year of the follow up after the removal of the ameloblastoma. There are some reports suggesting the pathologic potential of the pericoronal tissues of impacted third molars to develop odontogenic keratocysts and ameloblastomas. The current case reports a rare possibility of ameloblastic change at the site of uneventful healing after third molar extraction and orthognathic surgery.