Case Report
Copyright ©The Author(s) 2018. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Mar 7, 2018; 24(9): 1056-1062
Published online Mar 7, 2018. doi: 10.3748/wjg.v24.i9.1056
Four cancer cases after esophageal atresia repair: Time to start screening the upper gastrointestinal tract
Floor WT Vergouwe, Madeleine Gottrand, Bas PL Wijnhoven, Hanneke IJsselstijn, Guillaume Piessen, Marco J Bruno, René MH Wijnen, Manon CW Spaander
Floor WT Vergouwe, Marco J Bruno, Manon CW Spaander, Department of Gastroenterology and Hepatology, Erasmus MC University Medical Center, Rotterdam 3000 CA, Netherlands
Floor WT Vergouwe, Hanneke IJsselstijn, René MH Wijnen, Department of Pediatric Surgery, Erasmus MC University Medical Center - Sophia Children's Hospital, Rotterdam 3000 CB, Netherlands
Madeleine Gottrand, Department of Pediatrics, Jeanne de Flandre Children’s Hospital - Univ. Lille, CHU Lille, Lille 59000, France
Bas PL Wijnhoven, Department of Surgery, Erasmus MC University Medical Center, Rotterdam 3000 CA, Netherlands
Guillaume Piessen, Department of Digestive and Oncological Surgery, Claude Huriez Hospital - Univ. Lille, CHU Lille, Lille 59000, France
Author contributions: Vergouwe FWT, Gottrand M, Wijnhoven BPL and Piessen G collected clinical data; Vergouwe FWT, IJsselstijn H and Spaander MCW designed the manuscript; Vergouwe FWT, IJsselstijn H and Spaander MCW interpreted the data and drafted the manuscript; Gottrand M, Wijnhoven BPL, Piessen G, Bruno MJ and Wijnen RMH interpreted the data and critically revised the manuscript; all authors gave their final approval of the version of the article to be published.
Informed consent statement: Informed consent was waived since all handling to the subjects was part of standard clinical care.
Conflict-of-interest statement: All authors have no conflicts of interest to declare.
Open-Access: This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Correspondence to: Manon CW Spaander, MD, PhD, Associate Professor, Department of Gastroenterology and Hepatology, Erasmus MC University Medical Center, P.O. Box 2040, Rotterdam 3000 CA, Netherlands. v.spaander@erasmusmc.nl
Telephone: +31-10-7035643 Fax: +31-10-7035172
Received: October 26, 2017
Peer-review started: October 28, 2017
First decision: November 22, 2017
Revised: December 4, 2017
Accepted: January 24, 2018
Article in press: January 24, 2018
Published online: March 7, 2018
Processing time: 52 Days and 17.4 Hours
Abstract

Esophageal atresia (EA) is one of the most common congenital digestive malformations and requires surgical correction early in life. Dedicated centers have reported survival rates up to 95%. The most frequent comorbidities after EA repair are dysphagia (72%) and gastroesophageal reflux (GER) (67%). Chronic GER after EA repair might lead to mucosal damage, esophageal stricturing, Barrett’s esophagus and eventually esophageal adenocarcinoma. Several long-term follow-up studies found an increased risk of Barrett’s esophagus and esophageal carcinoma in EA patients, both at a relatively young age. Given these findings, the recent ESPGHAN-NASPGHAN guideline recommends routine endoscopy in adults born with EA. We report a series of four EA patients who developed a carcinoma of the gastrointestinal tract: three esophageal carcinoma and one colorectal carcinoma in a colonic interposition. These cases emphasize the importance of lifelong screening of the upper gastrointestinal tract in EA patients.

Keywords: Adenocarcinoma; Esophageal atresia; Esophageal cancer; Screening; Barrett’s esophagus; Squamous cell carcinoma

Core tip: Esophageal atresia (EA) is a common congenital malformation that requires surgical correction early in life. Improved perioperative care and surgical techniques have increased the survival rate. Gastroesophageal reflux and stasis are common after surgical repair and may be associated with an increased esophageal cancer risk. However, data on incidence and risk factors for esophageal carcinogenesis after EA repair are scarce. The recent ESPGHAN-NASPGHAN guideline recommends routine endoscopy in adults born with EA. Here we report four cancer cases at a relatively young age after EA repair: three esophageal carcinoma and one colorectal carcinoma in a colonic interposition.