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LETTER TO EDITOR
Year : 2007  |  Volume : 12  |  Issue : 1  |  Page : 58-59
 

Tracheo-bronchial remnants (chondroepithelial choristoma): An unusual cause of dysphagia


Dept. of Pathology, Grant Medical College and Sir. JJ Gr. of Hospital, Byculla, Mumbai - 8, Maharashatra, India

Correspondence Address:
Yoganand V Patil
Dept. of Pathology, Grant Medical College and Sir. JJ Gr. of Hospital, Byculla, Mumbai - 8, Maharashatra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0971-9261.31098

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How to cite this article:
D'Costa GF, Hastak MS, Sanklecha VM, Patil YV. Tracheo-bronchial remnants (chondroepithelial choristoma): An unusual cause of dysphagia. J Indian Assoc Pediatr Surg 2007;12:58-9

How to cite this URL:
D'Costa GF, Hastak MS, Sanklecha VM, Patil YV. Tracheo-bronchial remnants (chondroepithelial choristoma): An unusual cause of dysphagia. J Indian Assoc Pediatr Surg [serial online] 2007 [cited 2020 Oct 24];12:58-9. Available from: https://www.jiaps.com/text.asp?2007/12/1/58/31098


Sir,

Chondroepithelial choristoma also known as tracheo-bronchial remnants or cartilaginous esophageal rings is a rare cause of distal esophageal stenosis. Because of its rarity, the diagnosis is often overlooked in infants and young children presenting with feeding problems, especially when there are other major associated anomalies, and inappropriate treatment is often carried out before the correct diagnosis is established.

A 2-year-old female presented with vomiting immediately after food, the vomitus contained food particles and there was difficulty in swallowing solid food and inability to pass the Ryle's tube. A barium swallow showed a stricture at the lower end of the esophagus with proximal dilation. The esophagoscope could not be passed beyond the stricture, it showed inflammed esophagus with accumulation of food above the stricture. An esophageal resection of the stricture with anastomosis was done.

The etiology of this developmental anomaly is the defective separation of the embryonic respiratory tract from the primitive foregut resulting in sequestration of tracheo-bronchial precursor cells in the wall of the esophagus, which gets carried down during the growth process.[1] The other postulate is localized metaplasia of mesenchymal cells in the wall of the esophagus producing the cartilage and epithelial tissues of the respiratory tract.[1] Esophageal atresia occurs at the same embryonic stage in which the tracheal cartilage sequestrates in the esophageal wall. This explains the high incidence of the combination of esophageal atresia and stenosis due to tracheo-bronchial remnants.[2],[3] Anorectal anomalies are frequently associated with both these conditions.[2]

The symptoms start in early infancy and include recurrent vomiting, regurgitation of fluids and progressive dysphasia usually beginning with weaning. These symptoms suggest partial esophageal obstruction. Aspiration into the lung also occurs and there are recurrent bouts of respiratory distress and respiratory infections.[1],[8] The X-ray or barium swallow shows a localized constriction of the distal esophagus with proximal dilation.[3],[4],[5] Linear intramural clefts projecting horizontally from the area of stenosis correspond to the respiratory ducts.[1],[5],[6] The stenotic segment fails to relax on swallowing.

The differential diagnosis includes stenosis due to caustic agent ingestion, peptic esophagitis with or without hiatus hernia, achalasia cardia, esophageal webs and fibromuscular stenosis.[5] Our clinician's differential diagnosis was reflux stricture, congenital esophageal stricture and achalasia cardia. A resection anastomosis was done.

Grossly, there is a fusiform constriction of the involved segment <2 cm in length. Cut section reveals incomplete cartilaginous rings. We received multiple bits of tissue 2 x 2 x 2 cm, with unremarkable mucosa, wall uniformly thickened with small whitish nodules.

Histologically, the esophageal wall shows large or small, cartilaginous tracheal rings, submucosal and mucosal glands of tracheal origin, ducts lined by respiratory epithelium, surrounded by lymphoid tissue, some communicating with the lumen, rarely is heterotrophic pancreatic tissue seen.[3],[5],[7] Sometimes all the structures may not be seen. In our case only cartilage and tracheal bronchial glands were seen [Figure - 1],[Figure - 2],[Figure - 3].

The cause of obstruction may be mechanical, if the cartilaginous rings are large or interruption of progressive esophageal peristalsis, if they are small.[7] Arrangement of the respiratory elements could be in an orderly topographic manner, analogous to that occurring in the normal upper respiratory tract, as was in our case, where the name tracheo-bronchial remnant is more suitable.[8] If the lesion displays a loss of organized arrangement of its component elements with an intimate blending of glandular and cartilaginous structures, the term "chondroepithelial choristoma" is more appropriate.[8]

 
   References Top

1.Kumar R. A case of congenital oesophageal stricture due to a cartilaginous ring. Br J Surg 1962;49:533-4.  Back to cited text no. 1  [PUBMED]  
2.Nishina T, Tsuchida Y, Saito S. Congenital esophageal stenosis due to tracheobronchial remnants and its associated anomalies. J Pediatr Surg 1981;16:190-3.  Back to cited text no. 2  [PUBMED]  [FULLTEXT]
3.Zhao LL, Hsieh WS, Hsu WM. Congenital esophageal stenosis owing to ectopic tracheobronchial remnants. J Pediatr Surg 2004;39:1183-7.   Back to cited text no. 3  [PUBMED]  [FULLTEXT]
4.Yeung CK, Spitz L, Brereton RJ, Kiely EM, Leake J. Congenital esophageal stenosis due to tracheobronchial remnants: A rare but important association with esophageal atresia. J Pediatr Surg 1992;27:852-5.  Back to cited text no. 4  [PUBMED]  [FULLTEXT]
5.Rose JS, Kassner EG, Jurgens KH, Farman J. Congenital oesophageal strictures due to cartilaginous rings. Br J Radiol 1975;48:16-8.  Back to cited text no. 5  [PUBMED]  
6.Case 42411. Abortive congenital esophago-tracheal fistula. Case records of the Massachusetts general hospital. N Engl J Med 1956;255:707-10.  Back to cited text no. 6    
7.Ishida M, Tsuchida Y, Saito S, Tsunoda A. Congenital esophageal stenosis due to tracheobronchial remnants. J Pediatr Surg 1969;4:339-45.  Back to cited text no. 7  [PUBMED]  [FULLTEXT]
8.Goldman RL, Ban JL. Chondroepithelial choristoma (tracheobronchial rest) of the esophagus associated with esophageal atresia. Report of an unusual case. J Thorac Cardiovasc Surg 1972;63:318-21.  Back to cited text no. 8    


    Figures

[Figure - 1], [Figure - 2], [Figure - 3]



 

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