Tubular Esophageal Duplication

Abstract

Infected total tubular esophageal duplication was detected in an 11-year-old boy presenting with chest pain, dysphagia, cough, and fever. Esophagram (supplemental Fig. 1 [esophagram: water-soluble contrast medium showed a double esophageal lumen extending approximately 5 cm; arrowheads: double lumen, nonlinear arrow: septum, stars: communication between the 2 lumens, arrow: main lumen], https://links.lww.com/MPG/A161) and computed tomography (supplemental Fig. 2 [chest computed tomogram; minimum intensity projection on the sagittal plane confirmed a double esophageal lumen], https://links.lww.com/MPG/A162) revealed an esophageal pseudolumen. Endoscopy showed 2 esophageal lumens (Fig. 1). The main lumen was larger with normal mucosa, whereas the accessory lumen was narrower and lined with ulcerated tissue. The gastroscope was passed into the accessory lumen up to its distal communication with the main lumen. Surgery has been the unique treatment so far (1–3), except for a recently reported experience in operative endoscopy (4). An endoscopic treatment was therefore attempted. Using a standard videogastroscope, a guidewire was firstly placed in the accessory lumen, then a diathermic knife (ITDK, Lorenzatto, Turin, Italy), having an insulated ball in its tip to avoid potential injury beyond the cut zone (5), was inserted and connected to an intelligent electrosurgical workstation (ICC 200 E; ERBE, Marietta, GA). Starting from the upper end of the duplication and using the guidewire as reference for a straight incision, a lengthwise step-by-step mixed cutting and cautery of the intraluminal bridge were then performed (Fig. 2), with the ENDO-CUT mode set at 50/25 W, respectively. No complications were recorded. The child was discharged on day 3 after the procedure and is presently asymptomatic at 18-month follow-up. (supplemental Fig. 3 [endoscopy 3 months after the procedure; unique lumen with 2 longitudinal mucosal folds as scars of the previous incision of the intraluminal bridge], https://links.lww.com/MPG/A163).FIGURE 1: Upper endoscopy of the esophagus. 1: main lumen; 2: intraluminal bridge; 3:accessory lumen.FIGURE 2: Incision of the intraluminal bridge (2) using an ITDK (4). Superiorly the main lumen (1); inferiorly the accessory lumen (3).Esophageal duplications represent about 10% of all of the foregut duplications. The tubular type is extremely rare (5%–10% of cases) (1). They frequently appear before 2 years of age but can be incidentally discovered in adulthood (2,6). Most frequent symptoms are dysphagia, hemorrhage, chest pain, compression of adjacent organs, infection, perforation, and respiratory involvement. This observation confirms the possibility that total tubular esophageal duplication can be successfully managed by endoscopy avoiding surgery.