Two case reports of unexpected tracheal agenesis in the neonate: 3 C’s beyond algorithms for difficult airway management

Grass, Beate; Simma, Leopold; Reinehr, Michael; Zimmermann, Urs; Gysin, Claudine; Henze, Georg; Cannizzaro, Vincenzo

doi:10.1186/s12887-017-0806-z

Cited by 3 publications

(4 citation statements)

References 22 publications

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“…, ankylosing spondylitis, degenerative osteoarthritis, Treacher–Collins, Klippel–Feil, Down syndrome, mucopolysaccharidosis, and airway masses) (Category B4-H evidence ). 71–122…”

Section: Guidelinesmentioning

confidence: 99%

2022 American Society of Anesthesiologists Practice Guidelines for Management of the Difficult Airway

Apfelbaum¹,

Hagberg²,

Connis³

et al. 2021

Anesthesiology

602

547

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The American Society of Anesthesiologists; All India Difficult Airway Association; European Airway Management Society; European Society of Anaesthesiology and Intensive Care; Italian Society of Anesthesiology, Analgesia, Resuscitation and Intensive Care, Learning, Teaching and Investigation Difficult Airway Group; Society for Airway Management; Society for Ambulatory Anesthesia; Society for Head and Neck Anesthesia; Society for Pediatric Anesthesia; Society of Critical Care Anesthesiologists; and the Trauma Anesthesiology Society present an updated report of the Practice Guidelines for Management of the Difficult Airway.

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“…, ankylosing spondylitis, degenerative osteoarthritis, Treacher–Collins, Klippel–Feil, Down syndrome, mucopolysaccharidosis, and airway masses) (Category B4-H evidence ). 71–122…”

Section: Guidelinesmentioning

confidence: 99%

2022 American Society of Anesthesiologists Practice Guidelines for Management of the Difficult Airway

Apfelbaum¹,

Hagberg²,

Connis³

et al. 2021

Anesthesiology

602

547

View full text Add to dashboard Cite

show abstract

Section: Discussionmentioning

confidence: 99%

“…[ 1 , 3 , 19 , 20 ]. However, a TEF is frequently associated with TA and LA, making US diagnosis challenging as polyhydramnios is the single most common, though non-specific, prenatal finding [ 3 , 10 , 19 , 21 ]. Once other causes of polyhydramnios have been excluded and fetal airway obstruction is suspected, fetal MRI can confirm the diagnosis and locate the site of obstruction, and complete genetic evaluation is essential to rule out genetic anomalies and syndromes [ 3 , 4 , 19 , 22 ].…”

Section: Discussionmentioning

confidence: 99%

Neonatal Respiratory Distress and Airway Emergency: Report of Two Cases

et al. 2021

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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.

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“…Immediate survival requires a patent TEF and prompt realization of the need to ventilate through the native esophagus; teamwork, awareness, and communication are key components of survival. 20,21 Long-term management requires a multidisciplinary approach. While the literature paints a realistic picture of high mortality, recent reports suggest that TA survivors exist and highlight innovative techniques to improve survivors' lifespan and quality of life.…”

Section: Discussionmentioning

confidence: 99%

Tracheal Agenesis: Vertical Division of the Native Esophagus – A Novel Surgical Approach and Review of the Literature

Straughan

Mulcahy

Sandler

et al. 2020

Ann Otol Rhinol Laryngol

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Introduction: Tracheal agenesis (TA) is rare and usually fatal. Few survivors with concomitant tracheoesophageal fistulae (TEF) who underwent ligation of the distal esophagus with creation of a spit-fistula and neo-trachea from the proximal esophagus exist. We report a novel surgical technique whereby the esophagus is divided longitudinally to preserve a functional alimentation tract and a parallel neo-trachea. We review the literature of reported cases, including survivors beyond 12 months. Methods: Case report and literature review. Results: A female infant with prenatal polyhydramnios was born at 35 weeks gestation with immediate respiratory distress and absent cry. Oxygenation was maintained with a laryngeal mask airway. Despite a normal appearing larynx, she could not be intubated and emergent neck exploration disclosed no cervical trachea. The patient was placed on extra corporeal membranous oxygenation (ECMO), and later diagnosed with TA, Floyd Type I. Parental desire for reconstruction but refusal of a spit-fistula necessitated a novel procedure. The esophagus was divided longitudinally via a microstapler to preserve the original alimentary tract and create a parallel neo-trachea originating from the TEF and terminating as a cervical stoma. The healing process was complicated but the baby was ultimately discharged to home where she developed normally neurologically until succumbing one night to accidental decannulation at 16 months of age. Conclusion: We describe a novel surgical approach to manage TA. This includes avoiding creation of a spit fistula and preserving the native esophagus. We then survey the literature, reporting the survivorship duration and operative management of 174 reported cases of TA.

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Two case reports of unexpected tracheal agenesis in the neonate: 3 C’s beyond algorithms for difficult airway management

Cited by 3 publications

References 22 publications

2022 American Society of Anesthesiologists Practice Guidelines for Management of the Difficult Airway

2022 American Society of Anesthesiologists Practice Guidelines for Management of the Difficult Airway

Neonatal Respiratory Distress and Airway Emergency: Report of Two Cases

Tracheal Agenesis: Vertical Division of the Native Esophagus – A Novel Surgical Approach and Review of the Literature

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