Tracheal Atresia Associated with Cor Biloculare

McCracken, A. W.; Flanagan, N. G.; Flanagan, Paul

doi:10.1136/thx.19.6.530

Cited by 15 publications

(4 citation statements)

References 12 publications

(5 reference statements)

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“…Tracheal agenesis appears to have been described first by Payne [ 19001; subsequently 41 other patients were reported [Walcher, 1928;Fritz, 1933;Benesova and Peter, 1934;Marek, 1940;Morison, 1949;Milles and Dorsey, 1950;Kessel and Smith, 1953;Sandison, 1955;Hempel, 1956;Devenis and Otis, 1957;Onarir and Sariyalcin, 1961;Floyd et al, 1962;Witzleben, 1963;Fonkalsrud et al, 1963;McCracken et al, 1964;Devi and More, 1966;Stowens, 1966;Colavecchio, 1968;Lyons and Bruce, 1968;Eimer, 1969;Joshi, 1969;McNie and Pryse-Davies, 1970;Altman et al, 1972;Hopkinson, 1972;Hong et al, 1973;Effman et al, 1975;Chaurasia, 1976;Warfel, 1976;Donovan and Friedman, 1979;Far0 et al, 1979;Buchino et al, 1982; DFC:23 24 Evans, Reggin, and Greenberg Haupt and Edwards, 19831. In these cases tracheal agenesis was partial or total and various classifications of this malformation have been described [Floyd et al, 1962;Faro et al, 1979;Kluth, 19761.…”

Section: Pathogenesismentioning

confidence: 94%

Tracheal agenesis and associated malformations: A comparison with tracheoesophageal fistula and the VACTERL association

Reggin

Greenberg

1985

Am. J. Med. Genet.

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Tracheal agenesis is a rare malformation of the lower respiratory tract. Investigation of a patient with multiple congenital anomalies and tracheal agenesis prompted a review of the literature which uncovered 42 previously published cases, most of whom had other defects. The presence in our patient of a tracheal abnormality in association with radial hypoplasia, single umbilical artery, tetralogy of Fallot, and left hydroureter initially suggested presence of the VACTERL association. However, numerical classification of malformation patterns in the reported patients with tracheal agenesis and in a series of patients with tracheoesophageal fistula and other components of the VACTERL association suggests that tracheal agenesis does not occur in the VACTERL association and may be part of another pattern of malformations which includes laryngeal atresia, complex congenital heart anomalies, radial ray defects, and duodenal atresia.

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Section: Pathogenesismentioning

confidence: 94%

Tracheal agenesis and associated malformations: A comparison with tracheoesophageal fistula and the VACTERL association

Reggin

Greenberg

1985

Am. J. Med. Genet.

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“…The terms tracheal agenesis, tracheal atresia , and tracheal aplasia have each been used in the literature to refer to essentially the same entity 4–8 . These terms have also been used to differentiate more severe from less severe cases.…”

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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“…Atresia of the trachea, on the other hand, is extremely rare (Potter, 1952;Willis, 1962). McCracken, Flanagan and Flanagan (1964) collected 14 cases from the literature and added one of their own, and Lyons and Bruce (1968) added a further two cases.The present case is the 24th to be recorded and the eighth in the British literature. It presents features of developmental anatomy that differ from those of other cases and is the first case of obstruction of both the trachea and the oesophagus.…”

mentioning

confidence: 99%

mentioning

confidence: 99%