Purpose <p>Although the superior semicircular canal dehiscence syndrome was described at the end of the 20th century, we want to check if it is a pathology that has existed since ancient times, through the anthropological study of bone remains.</p> Methods <p>We have carried out an anthropological and radiological study (CT scan) of 8 skulls found in caves, as secondary burials of the Arbolí type epicampaniform culture (1800 − 1700 BC) on the Iberian Peninsula.</p> Results <p>The 8 skulls (16 temporal bones) show a grade 4 degree of pneumatisation or hyperpneumatization. One of these skulls, belonging to a male subject of around 25–30 years of age, shows a double dehiscence (superior semicircular canal and tegmen tympani) on the right side, and a possible congenital muscular torticollis on the same side.</p> Conclusion <p>Superior semicircular canal dehiscence syndrome already existed in an inhabitant from 1800 − 1700 BC (Iberian Peninsula). This is the first case in which the association of both dehiscences (superior semicircular canal and tegmen tympani) has been demonstrated.</p>