Agenesis of the trachea

Rovira, Joan Carles; Morales, Luis Antonio Ortiz; Rottermann, M.; Victoria, Julia; Llaurado, F; Púlgar, J. Pérez del

doi:10.1016/s0022-3468(89)80091-0

Cited by 10 publications

(6 citation statements)

References 6 publications

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“…The diagnosis of TA should be considered in a newborn with the triad of respiratory distress and difficulty in intubation, absent cry, and polyhydramnios [12,13]. Esophageal intubation in these infants may be lifesaving [13].…”

Section: Discussionmentioning

confidence: 99%

Tracheal agenesis with unique anatomy

Fraser

Stewart

Grant

et al. 2005

Journal of Pediatric Surgery

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Section: Discussionmentioning

confidence: 99%

Tracheal agenesis with unique anatomy

Fraser

Stewart

Grant

et al. 2005

Journal of Pediatric Surgery

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“…Other attempts have been made since, using essentially the same principles, and without any significant success. 36,57,68 The attempts at surgical correction are hampered by two major factors when the esophagus is used as a pseudotrachea. The first difficulty is that the esophagus lacks rigidity and is therefore prone to collapse with normal inspiratory forces.…”

Section: Discussionmentioning

confidence: 99%

Tracheal Agenesis: A Case Report and Review of the Literature

Kerschner

Klotch

1997

Otolaryngol.--head neck surg.

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Tracheal agenesis is a devastating congenital anomaly first described in 1900 by Payne. 1 In 1962 Floyd et al.2 classified tracheal agenesis into three types (Figs. 1, 2, and 3). One year later, Fonkalsrud et al. 3 attempted the first surgical correction of this anomaly. This attempt, as well as other interventions, have been unsuccessful in allowing any long-term survival.Eighty-seven cases have previously been identified in the world literature. This additional Floyd type III case typifies the problems of assessment and management of this fatal anomaly. A discussion of the embryology and associated congenital anomalies is presented. The clinical aspects of managing this abnormality are described along with previous attempts at surgical correction. Newer investigations may lead to a solution of this lethal anomaly. CASE REPORTA full-term, 3300-gm girl was delivered by normal spontaneous vaginal delivery to a 23-year-old gravida 2, para 1 mother who had an uncomplicated pregnancy with a normalappearing ultrasound at 20 weeks. After delivery the child was noted to have respiratory distress with Apgar scores of 3 and 6 at 1 and 5 minutes. The infant was noted to have an absent cry. Attempt was made to intubate the infant without success. Saturations were maintained with the use of bag-valve mask. Attempts at intubation by the neonatology service were also unsuccessful, and the child was taken to the neonatal intensive care unit while being supported by bag and mask. The ototaryngology service was called, and after direct laryngoscopy revealed no subglottic opening, the infant was taken directly to the operating room. Further endoscopy at this time revealed essentially normal-appearing vocal folds with complete occlusion of the airway immediately in the subglottic area. A patent esophagus was identified posteriorly. A neck exploration was performed in an attempt to identify a tracheal rem-From the Divisions of Otolaryngology (Dr.

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“…Initial literature searches disclosed an additional 66 cases not reviewed in our original paper [Smith and Bain, 1965;Reinhold-Richter, 1976;Perel et al, 1977;Jones and Drut, 1979;Morgan et al, 1979;Alfrey et al,1980;Anderson et al, 1981;Ohtani et al, 1982;Siplovich and Cywes, 1982;Parameswaran et al, 1983;Paes et al, 1984;Reilly et al, 1984;Solano-Larrañ aga and Coronado-Macías, 1984; Haskovcová , 1985;Milstein et al, 1985;Sakai et al, 1986;Holinger et al, 1987;Kearns and Miller, 1987;Spitz et al, 1987;Bray and Lamb, 1988;Esque et al, 1988;Herbert et al, 1988;Niranjan et al, 1988; Zimmerman et al, 1988;Arizmendi-Dorantes et al, 1989;Bradenkamp et al, 1989;Branger et al, 1989;Diaz et al,1989;Ishii et al, 1989;Nakada et al, 1989;Rovira et al, 1989;Scurry et al, 1989;Statz et al, 1989;Brichard et al, 1990;Chiu et al, 1990;McDonald and Oriol, 1990;Dost et al, 1991;Lopez et al, 1991; Schiffman et al, 1991;Weil, 1991;Coronel et al, 1992;…”

Section: Methodsmentioning

confidence: 97%

Tracheal agenesis revisited: analysis of associated anomalies

1999

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We describe five new cases of tracheal agenesis and report on epidemiological and numerical analyses of nearly 100 such cases with multiple congenital anomalies. Malformations seen with tracheal agenesis form patterns which overlap with, but are distinct from, VACTERL association. They have a high frequency of other lower respiratory tract anomalies; e.g., laryngeal atresia and lung lobation defects, and complex heart anomalies, but fewer anal and vertebral malformations. Cluster analysis of the malformations in 86 patients identified four consistent groups. Anomalies in the first group were primarily restricted to the trachea, larynx, and cardiovascular system. In the second group, the patients had more severe cardiac defects, and lung lobation anomalies, while in the third they had a caudal component in addition to thoracic abnormalities, with anal and renal anomalies being common. Each of these groups showed a male excess and may represent increasingly severe perturbations in development fields encompassing the developing respiratory tract. Although the nature of the causative insult is unknown and probably heterogenous, one underlying pathogenetic mechanism may be abnormal epithelial-mesenchymal interactions. Patients in the fourth group also had multisystem involvement with a high incidence of aberrant vessels, complex cardiac malformations, lung lobation defects, and anomalies of other foregut derivatives. The sex ratio in this group was normal and such cases could represent a disturbance in the primary development field during blastogenesis with secondary vascular disruptions. Complete tracheal agenesis is a lethal anomaly. However, segmental forms may be correctable and, in this group of infants, the nature of associated anomalies may well determine long-term prognosis.

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Agenesis of the trachea

Cited by 10 publications

References 6 publications

Tracheal agenesis with unique anatomy

Tracheal agenesis with unique anatomy

Tracheal Agenesis: A Case Report and Review of the Literature

Tracheal agenesis revisited: analysis of associated anomalies

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