Odontogenic keratocysts, characterized by considerable local aggressiveness and high recurrence rates, account for 5-15% of all odontogenic cysts. This paper reviews the literature and describes the most important aspects of this type of cyst, a clinical example being given as way of illustration. We also report a paleopathological case featuring an osteolytic lesion where the characteristics are consistent with a keratocyst. The aim is thus to encourage odontologists to contribute to research in the field of paleopathology.