Rare type of tracheal agenesis: Unexpected presentation and immediate consideration of emergent esophageal intubation in neonatal resuscitation program. Case reports and review of the literature

• Published in: Pediatric pulmonology Vol. 59; no. 6; pp. 1757 - 1764
• Main Authors: Wu, Yi‐Hsin, Hsiao, Ching‐Heng, Chen, Yi‐Ling, Tsai, Li‐Yi, Mu, Shu‐Chi
• Format: Journal Article
• Language: English
• Published: United States Wiley Subscription Services, Inc 01.06.2024
• Subjects: airway malformation; Cardiopulmonary resuscitation; Case reports; congenital anomaly; Constriction, Pathologic; CPR; Cyanosis; Cyanosis - etiology; Esophagus; Esophagus - abnormalities; Esophagus - diagnostic imaging; Female; Humans; Infant, Newborn; Intubation; Intubation, Intratracheal - methods; Literature reviews; Male; Neonatal care; newborn; Resuscitation - methods; Trachea - abnormalities; Trachea - diagnostic imaging; Tracheoesophageal Fistula - complications; Tracheoesophageal Fistula - surgery; voiceless crying; newborn; voiceless crying; congenital anomaly; airway malformation; cyanosis
• ISSN: 8755-6863; 1099-0496; 1099-0496
• DOI: 10.1002/ppul.26990

Background Tracheal agenesis, or tracheal atresia, is a rare congenital anomaly. The presence of a tracheoesophageal fistula (TEF) can help with breathing for newborns with tracheal agenesis. In this article, we presented three unique cases and outcomes of neonates with tracheal agenesis along with a review of the literature. Methods This study consisted of a single center case series followed by a review of literature. Case reports were generated using both written and electronic medical records from a single hospital. We summarized three unique cases and outcomes of neonates with tracheal agenesis and performed a review of the literature. Results We identified three cases of tracheal agenesis presented with severe cyanosis without spontaneous crying upon birth. Experienced pediatricians attempted to intubate the babies but were unsuccessful. Endotracheal tubes were subsequently either accidentally or purposely placed into the esophagus, and oxygen saturation levels improved. This suggested tracheal agenesis with TEF. Two cases underwent surgical intervention after resuscitation with esophageal intubation. Conclusion Esophageal intubation may be a life‐sustaining ventilation support for patients with tracheal agenesis and TEF at initial resuscitation. Clinicians should suspect tracheal agenesis when a newborn presents with severe cyanosis and voiceless crying upon birth, and esophageal intubation should be immediately attempted.