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

Author:

Wu Yi‐Hsin1,Hsiao Ching‐Heng23ORCID,Chen Yi‐Ling1,Tsai Li‐Yi1,Mu Shu‐Chi14

Affiliation:

1. Department of Pediatrics Shin‐Kong Wu Ho‐Su Memorial Hospital Taipei Taiwan

2. Department of Medical Education Taichung Veterans General Hospital Taichung Taiwan

3. School of Medicine, College of Medicine Taipei Medical University Taipei City Taiwan

4. School of Medicine, College of Medicine Fu Jen Catholic University New Taipei City Taiwan

Abstract

AbstractBackgroundTracheal 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.MethodsThis 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.ResultsWe 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.ConclusionEsophageal 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.

Publisher

Wiley

Reference26 articles.

1. Tracheal agenesis: a rare but fatal congenital anomaly;Ergun S;Mcgill J Med,2011

2. Tracheal agenesis1 1No competing interests declared.

3. Tracheal agenesis revisited: analysis of associated anomalies

4. Congenital absence of the trachea;Payne W;Brooklyn med J,1900

5. Agenesis of the trachea;Floyd J;Am Rev Respir Dis,1962

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