Neonatal Respiratory Distress and Airway Emergency: Report of Two Cases

Lorenzo Bresciani; Paola Grazioli; Roberta Bosio; Gaetano Chirico; Cesare Zambelloni; Amerigo Santoro; Carla Baronchelli; Luca O Redaelli de Zinis

doi:10.3390/children8040255

Neonatal Respiratory Distress and Airway Emergency: Report of Two Cases

Children (Basel). 2021 Mar 25;8(4):255. doi: 10.3390/children8040255.

Authors

Lorenzo Bresciani¹, Paola Grazioli¹, Roberta Bosio¹, Gaetano Chirico², Cesare Zambelloni², Amerigo Santoro³, Carla Baronchelli³, Luca O Redaelli de Zinis^{1

4}

Affiliations

¹ Pediatric Otolaryngology Head Neck Surgery, Children Hospital "ASST Spedali Civili", 25123 Brescia, Italy.
² Department of Neonatology and Neonatal Intensive Care Unit, Children Hospital "ASST Spedali Civili", 25123 Brescia, Italy.
³ Department of Pathology, University of Brescia, 25123 Brescia, Italy.
⁴ Department of Medical and Surgical Specialties, Radiological Sciences and Public Health, Section of Audiology, University of Brescia, 25123 Brescia, Italy.

Abstract

We discuss two cases of congenital airway malformations seen in our neonatal intensive care unit (NICU). The aim is to report extremely rare events characterized by immediate respiratory distress after delivery and the impossibility to ventilate and intubate the airway. The first case is a male twin born at 34 weeks by emergency caesarean section. Immediately after delivery, the newborn was cyanotic and showed severe respiratory distress. Bag-valve-mask ventilation did not relieve the respiratory distress but allowed for temporary oxygenation during subsequent unsuccessful oral-tracheal intubation (OTI) attempts. Flexible laryngoscopy revealed complete subglottic obstruction. Postmortem analysis revealed a poly-malformative syndrome, unilateral multicystic renal dysplasia with a complete subglottic diaphragm, and a tracheo-esophageal fistula (TEF). The second case is a male patient that was vaginally born at 35 weeks. Antenatally, an ultrasound (US) arose suspicion for a VACTERL association (vertebral defects, anal atresia, TEF with esophageal atresia and radial or renal dysplasia, plus cardiovascular and limb defects) and a TEF, and thus, fetal magnetic resonance (MRI) was scheduled. Spontaneous labor started shortly thereafter, before imaging could be performed. Respiratory distress, cyanosis, and absence of an audible cry was observed immediately at delivery. Attempts at OTI were unsuccessful, whereas bag-valve-mask ventilation and esophageal intubation allowed for sufficient oxygenation. An emergency tracheostomy was attempted, although no trachea could be found on cervical exploration. Postmortem analysis revealed tracheal agenesis (TA), renal dysplasia, anal atresia, and a single umbilical artery. Clinicians need to be aware of congenital airway malformations and subsequent difficulties upon endotracheal intubation and must plan for multidisciplinary management of the airway at delivery, including emergency esophageal intubation and tracheostomy.

Keywords: CHAOS syndrome; congenital anomalies; intubation; tracheal agenesis.

Publication types

Case Reports