Partial craniofacial duplication in a resource-limited setting: a case report

Diprosopus, or craniofacial duplication, is an extremely rare congenital malformation, representing only 0.4% of conjoined twins with an estimated prevalence of 1 in 15 million births. Fewer than 40 cases have been reported worldwide. We describe a case of partial craniofacial duplication in a child from rural Ethiopia, highlighting the challenges of delayed diagnosis and management in a resource-limited setting.

A 7-year-old girl presented with a congenital mandibular mass that had remained untreated since birth due to a lack of antenatal care (ANC) and delayed health-seeking. Examination revealed a pedunculated soft tissue lesion containing duplicated rudimentary facial elements, including tooth-like structures and lip-like tissue. Laboratory investigations were normal, and skull radiography showed no bony abnormalities. Advanced imaging was not feasible. The mass was excised surgically, and gross and histopathological evaluation confirmed partial craniofacial duplication. Postoperative recovery was uneventful, with improved psychosocial outcomes.

Unlike complete diprosopus, which is typically incompatible with life, partial forms may permit long-term survival. The embryopathogenesis remains debated, with proposed mechanisms including incomplete embryonic disc division and neural crest duplication. While management in well-resourced settings is often facilitated by advanced imaging and multidisciplinary planning, this case demonstrates that direct surgical excision can achieve favorable outcomes even where resources are limited.

This rare case contributes to the limited literature on diprosopus and underscores the importance of strengthening ANC, anomaly screening, and community awareness. Timely surgical intervention, even in low-resource environments, can restore function and enhance psychosocial outcomes.