Congenital complete absence of tracheal rings with trifurcate carina: Case report of a rare clinical and endoscopic presentation

Schwartz, Isaac; Utz, Edward R.; Gaudreau, Philip

doi:10.1016/j.ijporl.2018.05.009

Cited by 3 publications

(4 citation statements)

References 11 publications

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“…According to existing literature, most of the children required treatment; only one case with mild symptoms did not undergo interventional treatment (6). Four patients in our study underwent surgery, and three of them recovered well; one died of severe infection and anastomotic dehiscence.…”

Section: Discussionmentioning

confidence: 66%

“…Tracheal cartilage deficiency may be located in the cervical, middle, and distal trachea with different lengths. Among them, the distal trachea seems to be the most common location (1,(3)(4)(5)(6)(7)(8). Histological examinations of the resected trachea show a cartilaginous islet or asymmetric, irregularly disconnected cartilaginous rings.…”

Section: Discussionmentioning

confidence: 99%

“…Six cases had short localized stenosis, one case was of distal tracheal stenosis and tracheal collapse, and five cases were of segmental tracheal collapse. Besides that, trifurcate carina was found in half of the cases (1,(4)(5)(6). Six cases of localized stenosis included one cervical tracheal stenosis, one middle tracheal stenosis, three distal tracheal stenoses proximal to the carina, and one in the juxtacarina location (5,7,8).…”

Section: Literature Reviewmentioning

confidence: 94%

“…Repeated balloon dilatations were required due to restenosis; cycled antibiotics and respiratory physiotherapy were prescribed due to severe bilateral bronchiectasis (1). The other patient, who was diagnosed with isolated deficient cartilage rings, survived without treatment, manifesting quiet stridor with normal development at 2 years of age (6). In three patients with segmental tracheal collapse, the middle and the distal trachea were involved; those patients were misdiagnosed with tracheomalacia in infancy.…”

Section: Literature Reviewmentioning

confidence: 99%

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Clinical Analysis of Congenital Deficient Tracheal Cartilage Rings: Six Case Reports and a Literature Review

Meng

Feng

et al. 2020

Front. Pediatr.

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Congenital deficiency of tracheal rings is a rare tracheal malformation that can cause central airway obstruction. Herein we reported the clinical data of six patients with symptomatic congenital deficient tracheal rings. There were five cases, with isolated short-segment absent cartilage ring located on the distal trachea (three cases), cervical trachea (one case), and distal trachea combined with bilateral bronchi (one case). Among them, four (4/5) received surgical tracheal resection, three fully recovered, and one died of severe infection. Besides that, one patient, who could not be weaned off the mechanical ventilation, died after rejecting surgery. One case had episodes of recurrent dyspnea and extubation failure due to long-segment tracheomalacia after repair of esophageal atresia and tracheoesophageal fistula. For this patient, deficient cartilage rings were suspected and confirmed at the age of 26 months. Moreover, the clinical characteristics of 12 cases with congenital deficient tracheal cartilage rings reported in previous literature were reviewed. The different characteristics between short-and long-segment deficient cartilage rings were discussed.

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Section: Discussionmentioning

confidence: 66%