Abstract

Objective: Esophageal duplications are very rare congenital malformations that can be diagnosed at any age. Patients remain asymptomatic unless complications occur. Surgery is the standard treatment. The experience of endoscopic treatment is very limited. We report a case of a boy with esophageal tubular duplication successfully treated with endoscopic resection. Case Report: A 10-year-old boy with an uneventful medical history was urgently admitted in a peripheral hospital for acute dysphagia and severe retrosternal pain. During admission sialorrhea, cough, and fever appeared. Physical examination and chest X-rays were normal. Antibiotics were started with fever and cough resolution, but persistence of severe dysphagia with weight loss of 7 kg. Endoscopy demonstrated two ulcerations in proximal and distal esophagus, with mucus and pus. After a chest CT scan an esophageal duplication was suspected. The patient was then referred to our Institute. Because our initial suspect was a dissecting intramural hematoma, the child was maintained fasted in parenteral nutrition and antibiotics were continued. Barium esophagram revealed a double-barrelled esophagus with a longitudinal septum between two lumens, suggesting intramural esophageal dissection or esophageal duplication. Upper endoscopy showed two orifices in the mid-esophagus. Endoscope (5.9 mm ∅, GIF-XP160, Olympus) advanced through the main lumen and then through the 5-cm-long pseudo-lumen that presented normal esophageal mucosa with partial ulcerations and a distal wide communication to the main one. A thick bridge separated the two lumens. The diagnosis of a complete tubular duplication was confirmed. A nasogastric tube was inserted in the main lumen under direct vision and enteral feeding was started. Therapeutic endoscopy under general anesthesia was finally performed, with paediatric surgeons in attendance in case of failure or complications. After the insertion of a guide wire in the pseudo-lumen an insulated tip diathermic knife (ITDK, M.G. Lorenzatto, Turin, Italy) was connected through the endoscope channel to an electrosurgical intelligent workstation (ICC 200 E, ERBE, GA, USA), setting the ENDO-CUT® mode at 50/25 W. Starting from the upper opening a step by step mixed cutting and cautery of the septum was done, obtaining a lengthwise resection. The endoscopic procedure was completed uneventfully with success. The patient started to drink in the same day and oral food intake was introduced the next day without problems. He was discharged within 72 hours. Repeated endoscopy after 4 months showed normal esophagus with 2 longitudinal residual folds, without stenosis. At 8 months follow-up he is asymptomatic and in excellent general condition with full weight recovery. Conclusions: The diagnosis of esophageal tubular duplication is often occasionally and not easy, especially during complications. Repeated upper endoscopy is sometimes necessary to clarify the etiology of esophageal wall abnormalities. The definitive treatment is surgical excision, although the endoscopic treatment has been recently suggested. To our knowledge, only one complete endoscopic management of a tubular esophageal duplication has been reported so far. We describe another case of successfully endoscopic resection without complications. We used an ITDK, a device particularly useful to avoid potential injury of esophageal wall during the cutting of the interluminal septum. Our experience adds information about operative endoscopy for these rare esophageal congenital defects. Expert endoscopist should consider this less-invasive method as a valid alternative to surgery. PP24

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