Oral cleft (OC) is a common congenital anomaly in humans, which occurs in approximately one per 700 live births. However, cleft skulls are found relatively seldom in archaeological specimens, especially for the bc period. Mortality of infants with OC was apparently high because of breastfeeding difficulties and culturally sanctioned rejection of them in the past. This paper presents one of the most ancient cases of OC in Eurasia. The finding comes from the Middle Bronze Age pastoralists' cemetery in Altai, Russia. The skeletal remains have been radiocarbon dated to 1883–1665 cal bc. Cranial and dental measurements, computed tomography and radiography have been used to examine this malformation. The facial sculpting reconstruction has been performed to visualise the ante mortem appearance of the cleft face. Besides, δ13C and δ15N isotope analysis was used for a comparative study of the diet of the cleft individual. It has been established that a young man from Altai was affected by complete bilateral cleft lip and alveolus, which is a very rare form of typical OC. This defect is associated with dental anomalies such as hypodontia, microdontia, and ectopia as well as with dysmorphology of the facial complex outside the cleft area, including mild hypertelorism, hypoplasia of nasal bones and antheroposterior maxillary deficiency. Furthermore, aplasia of the frontal sinuses and the sacral spina bifida occulta have been observed. These findings are consistent with clinical data indicating strong correlation between development of a major defect and multiple minor anomalies. The results of the study also suggest that there was tolerant attitude towards the facial deformities among the ancient Altai pastoralists. Copyright © 2016 John Wiley & Sons, Ltd.