A clinicopathological study of 57 unicystic ameloblastomas has been undertaken, which represents 15% of all cases of ameloblastoma accessioned in our department over a 30-yr period. Of the cases where gender was recorded: 30 were male and 23 female. The majority of patients were black (51 cases) and most occurred in the mandible (52). This distribution conforms to that of solid and multicystic ameloblastomas. The mean age at diagnosis was 23.8 years (S.D. 14.9) which is significantly younger than for the conventional counterpart (p less than 0.1%). The lesions were classified histologically into 3 groups: Group 1 (42%) cyst lined by a variable often non-descript epithelium; Group 2 (9%) cyst showing intraluminal plexiform proliferation of epithelium; Group 3 (49%) cyst with invasion of epithelium into the cyst wall in either follicular or plexiform patterns. While Group 1 and 2 lesions may be treated by enucleation, Group 3 lesions should be treated aggressively as for conventional ameloblastomas. The objectives of correct histological diagnosis, subclassification and appropriate therapy are best achieved by enucleation biopsy. There is little evidence to support origin from pre-existing odontogenic cysts.