We report a full-term male neonate found to have undiagnosed syngnathia requiring extensive resuscitation at birth followed by urgent tracheostomy. We conducted a systematic literature review to study the presentation, resuscitation methods, and outcomes of neonates with congenital syngnathia. Of the 174 cases reported to date, 91 had initial resuscitation data available. Extensive resuscitation was required in 16 of these 91 infants (18%). This ranged from nasal intubation to emergent tracheostomy. These neonates are potentially higher risk deliveries for which methods in addition to those recommended by the American Heart Association neonatal resuscitation guidelines may be needed.
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