Keratocystic odontogenic tumours raise particular interest, because of their high recurrence rate and association with nevoid basal cell carcinoma syndrome. OBJECTIVE: To analyze the clinical and histopathological features of all cases diagnosed as keratocystic odontogenic tumour in a Brazilian population. METHODS: A total of 64 keratocystic odontogenic tumours, arising in forty-six patients, were evaluated using the following parameters: association with nevoid basal cell carcinoma syndrome, gender, age at first diagnosis, race, anatomical location, symptoms, radiographic features, history of recurrence, association with teeth, and treatment. RESULTS: Keratocystic odontogenic tumours were more frequent among women than men (1:0.84). The mean patient age was 31.5 years (SD: +/- 16.6). Ten tumours (16.4%) involved the maxilla and 51 (83.6%) the mandible. Swelling (n = 12; 46.1%), followed by pain and swelling (n = 4; 15.3%), were most common clinical manifestations. A unilocular radiotransparency with well-defined margins was the main radiographic finding (n = 29; 87.8%). A significant association was observed between the multilocular radiographic pattern and recurrence (p < 0.05, Fisher's Test). Sixty-one (95.3%) tumours were treated by surgical enucleation followed by bone curettage, and the recurrence rate was 13% (n = 6). This study showed that the keratocystic odontogenic tumours relapsed within a mean period of 25-36 months. CONCLUSION: Despite the results of this study being similar to previous reports found in the literature, it provides an important insight about keratocystic odontogenic tumours in a Brazilian population