This paper discusses the differential diagnosis of an unusual bone tumour observed in the right maxilla of one adult calvarium, from the Late Neolithic collective burial of Lapa do Bugio (Sesimbra, Portugal). The differential diagnosis of the lesion gave rise to several possible pathological conditions, namely, ossifying fibroma, fibrous dysplasia, and focal cemento‐osseous dysplasia. Despite the impossibility of performing histological analysis, various macroscopic and radiological aspects lead us to consider ossifying fibroma as the most probable diagnose. This appears to be the earliest reported case of this condition in the osteoarchaeological literature, and adds to the very few palaeopathological case descriptions available for ossifying fibroma. Copyright © 2009 John Wiley & Sons, Ltd.