We present a 1500-year-old skull of a 5-year-old child (ossification and tooth eruption), who presented a premaxillary agenesis and ocular hypotelorism suggesting the diagnosis of semilobar HPE. The presentation of HPE has been extensively studied by Cohen (2009). He describes cyclopia as the main manifestation. 4 Although the presence of HPE and premaxillary agenesis has been reported, this case corresponds to the Lima culture, a pre-Inca culture that developed in the central coast of Peru (between A.D. 100 to A.D. 650). In our case it was not possible to determine the aetiology, considering that it was an indigenous community that inhabited Peru 1500 years ago. Different representations of human malformations in community-made ceramics South American Pacific coast with ornaments and costumes of the elite, suggest that people with congenital malformations had some preferential management in these communities. 5