Partial facial duplication (diprosopus): a  case report and review of the literature

• Published in: Journal of medical case reports Vol. 18; no. 1; p. 176
• Main Authors: Salah, Fathia Omer, Zewdie, Yohannes Girma, Ambachew, Semienew, Nour, Amal Saleh, Endale, Tewodros
• Format: Journal Article
• Language: English
• Published: England BioMed Central Ltd 23.03.2024; BioMed Central; BMC
• Subjects: Anencephaly; Blood; Blood tests; Case reports; Conjoined twins; Coronary vessels; Corpus callosum agenesis; Craniofacial duplication; CT imaging; Diprosopus; Face - abnormalities; Female; Humans; Infant, Newborn; Interhemispheric lipoma; Literature reviews; Magnetic resonance imaging; Mandible; Maxilla; Medical examination; Medical prognosis; Mouth; Neonatology; Nose; Obstetrics; Pneumonia; Pregnancy; Pregnant women; Sepsis; Ultrasonic imaging; Diprosopus; Interhemispheric lipoma; Craniofacial duplication; Corpus callosum agenesis
• ISSN: 1752-1947; 1752-1947
• DOI: 10.1186/s13256-024-04423-4

Diprosopus, or craniofacial duplication, is a rare entity that occurs in approximately 1 in 180,000 to 15 million live births. The degree of duplication varies from complete facial duplication to small facial structure duplication like the nose and eye. The cause of diprosopus is unknown though there are proposed factors. Our African patient was a term 72 hours old female neonate who was referred to our center with impression of lower facial duplication with two oral cavity that are located side to side separated by large soft tissue, she also had flat nasal bridge with widely separated nostrils and widely spaced eyes. Besides the facial malformation she had multiple episodes of vomiting with aspiration. Her blood tests were normal. Precontract brain computed tomography (CT) scan confirmed partially duplicated mandible and maxilla, two oral cavity separated by large fatty tissue, brain tissue were well formed and the only abnormality was corpus callosum agenesis and interhemispheric lipoma. In her stay at hospital nasogastric tube (NG) tube feed was initiated and started with antibiotics for aspiration pneumonia. After 25th day the neonatal passed away with possible cause of death being respiratory failure. Craniofacial duplication is a very rare anomaly with only a few cases reported. Most of these patients are stillborn, even if they survive the prognosis is often poor. Early prenatal diagnosis is very important as termination of pregnancy can sometimes be considered an option.