Bronchogenic cysts and esophageal duplications: common origins and treatment

K K Nobuhara, Y C Gorski, M P La Quaglia, R C Shamberger
Journal of Pediatric Surgery 1997, 32 (10): 1408-13

BACKGROUND/PURPOSE: Bronchogenic cysts and esophageal duplications are usually considered as separate foregut malformations. Yet, both are thought to arise from the same embryological event, division of the embryonic foregut, and they share common histological characteristics, often making their clinical differentiation difficult.

METHODS: A retrospective review of the cases of 68 children treated at a single institution between 1937 and 1995 was performed. Thirty children were girls (44%) and 38 were boys (56%). Ages ranged from newborn to 24 years. Complete records were available in all children. Fourteen of these 68 children were asymptomatic.

RESULTS: Respiratory (54%) or gastrointestinal (13%) symptoms were the most frequent presenting problems. The majority of children were treated by resection of the cyst (52 of 68; 76%), while 9 of 68 (13%) required lobectomy for intraparenchymal lesions. Three children underwent marsupialization, with all of these children requiring additional surgery for recurrent disease. Five children (5 of 68; 7%) had multiple cysts. The mortality rate from this series was 10% (7 of 68). Two deaths were caused by perioperative exsanguination, one related to bleeding from a cyst lined with gastric mucosa with subsequent ulceration and hemorrhage into the esophagus. Two deaths occurred secondary to septic complications, one from an esophageal leak and the other from an intraparenchymal abscess. Two deaths were caused by respiratory failure; one was unrelated (SIDS). The majority of cysts found on histological review were lined by respiratory epithelium or bronchial glands (51 of 68; 75%). Gastrointestinal epithelium was present in cysts of nine children, only two of which were clinically diagnosed as esophageal duplications. Twenty-one cases (21 of 68; 31%) were classified as esophageal duplications based on the intramural location of the cyst, yet 15 of 21 (71%) contained respiratory epithelium, substantiating the hypothesis of the common origin of these lesions.

CONCLUSIONS: The histological similarity and anatomic proximity of the "bronchogenic cysts" and the intramural "esophageal duplications" supports their common origin. The possible complications of bleeding, ulceration, infection, and obstruction of the esophagus or airway, should generally lead to prompt resection.

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