Neonatal esophageal trachealization and esophagocarinoplasty in the treatment of flow-limited Floyd II tracheal agenesis

Densmore, John C.; Oldham, Keith T.; Dominguez, Kathleen M.; Berdan, Elizabeth A.; McCormick, Michael E.; Beste, David J.; Amos, Louella B.; Lång, Cecilia; Woods, Ronald K.; Kouretas, Peter C.; Mitchell, Michael E.

doi:10.1016/j.jtcvs.2017.01.029

Cited by 19 publications

(25 citation statements)

References 25 publications

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“…Those few patients who survive postnatally require the presence of a functioning TEF that allows lifesaving esophageal intubation, followed by prompt distal esophageal occlusion and gastric decompression to allow for efficient ventilation. Subsequently, distal esophagostomy and esophageal neotrachealization or reconstruction is required [3,13,24,26]. Tazuke et al, in 2015, described four patients with TA with long-term survival (77-109 months) after airway and esophageal reconstruction [13].…”

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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Section: Discussionmentioning

confidence: 99%

Neonatal Respiratory Distress and Airway Emergency: Report of Two Cases

et al. 2021

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“…Park et al in 2015 reported survival up to almost 1 year for a type 2 TA while in the same year Tazuke et al reported survival ranging from 77 to 109 months for four children (two type 1, one type 2, one type 3) 15 16. Recently, Densmore et al reported survival (follow-up 146 days) in type 2 TA with neonatal trachealisation and oesophago-carinoplasty 17. Overall neo trachealisation of the oesophagus has been the surgery of choice; however, the outcomes still remain dismal for this rare condition.…”

Section: Discussionmentioning

confidence: 99%

“…Options in the future include tissue engineering offering a hope towards tracheal transplantation. Translational research and animal experiments may offer directions to a possibility of an ideal tracheal replacement with required rigidity, humidification, ciliary function and options of growth with the child 17. Till then TA remains a potentially lethal condition which can be a major burden to any healthcare system.…”

Section: Discussionmentioning

confidence: 99%

Tracheal agenesis in a new born: lessons learnt

Naina¹,

John

Kathar

et al. 2018

BMJ Case Reports

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Tracheal agenesis is a rare but fatal congenital tracheal malformation. Lack of prenatal symptom and a typical clinical presentation lead to failure to arrive at a correct diagnosis and confusion during resuscitation. We report a case of a newborn male child with type 2 tracheal agenesis. Despite a typical presentation, diagnosis was delayed after unsuccessful intubation, examination under anaesthesia and emergency tracheostomy. The embryology, diagnostic criteria and potential treatment options are discussed. This case report is valuable in increasing awareness of this rare condition and will help us in being better prepared in managing these children. Future studies should aim to find the optimal replacement for the tracheal.

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“…This type of tracheal agenesis and its surgical management is the subject of the case report in this issue of the Journal by Densmore and colleagues. 3 Salvage treatment of a critically ill neonate with tracheal agenesis is contingent on the presence of a tracheoesophageal or bronchoesophageal fistula, which allows esophageal intubation and mechanical ventilation. Prenatal diagnosis is rare and only suspected in the absence of a tracheoesophageal fistula.…”

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confidence: 99%

“…In 4 survivors reported from Osaka University, gastrointestinal continuity was established by gastric or jejunal interposition. 5 Densmore and colleagues 3 have modified this surgical approach to save a particularly high-risk patient who had a nearly atretic tracheoesophageal fistula and concerns for neurologic insult from postnatal hypoxia. Their report speaks to the coordinated effort required of multiple medical specialists, and the resources required at a tertiary care facility, to achieve a truly remarkable outcome for the patient and family.…”

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confidence: 99%