Observational Study
Copyright ©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastrointest Endosc. Sep 16, 2023; 15(9): 553-563
Published online Sep 16, 2023. doi: 10.4253/wjge.v15.i9.553
Graft dilatation and Barrett’s esophagus in adults after gastric pull-up and jejunal interposition for long-gap esophageal atresia
Eleonora Sofie van Tuyll van Serooskerken, Gabriele Gallo, Bas L Weusten, Jessie Westerhof, Lodewijk AA Brosens, Sander Zwaveling, Jetske Ruiterkamp, Jan BF Hulscher, Hubertus GM Arets, Arnold JN Bittermann, David C van der Zee, Stefaan HAJ Tytgat, Maud YA Lindeboom
Eleonora Sofie van Tuyll van Serooskerken, Jetske Ruiterkamp, David C van der Zee, Stefaan HAJ Tytgat, Maud YA Lindeboom, Department of Pediatric Surgery, Wilhelmina Children’s Hospital, Utrecht 3508 AB, Netherlands
Gabriele Gallo, Jan BF Hulscher, Department of Pediatric Surgery, University Medical Center Groningen, Groningen 9713 GZ, Netherlands
Bas L Weusten, Department of Gastroenterology and Hepatology, University Medical Center Utrecht, Utrecht 3508 AB, Netherlands
Jessie Westerhof, Department of Gastroenterology and Hepatology, University Medical Center Groningen, Groningen 9713 GZ, Netherlands
Lodewijk AA Brosens, Department of Pathology, University Medical Center Utrecht, Utrecht 3508 AB, Netherlands
Sander Zwaveling, Department of Pediatric Surgery, Amsterdam University Medical Center, Amsterdam 1105 AZ, Netherlands
Hubertus GM Arets, Department of Pediatric Pulmonology, Wilhelmina Children’s Hospital, Utrecht 3508 AB, Netherlands
Arnold JN Bittermann, Department of Pediatric Otorhinolaryngology, Wilhelmina Children’s Hospital, Utrecht 3508 AB, Netherlands
Author contributions: van Tuyll van Serooskerken ES, Gallo G, Hulscher JB, Tytgat SH, and Lindeboom MY conceptualized and designed the study; van Tuyll van Serooskerken ES and Gallo G collected the data, carried out the initial analysis, drafted the initial manuscript; van Tuyll van Serooskerken ES, Gallo G, Weusten BL, Westerhof J, Brosens LA, Zwaveling S, Ruiterkamp J, Hulscher JB, Arets HG, Bittermann AJ, van der Zee DC, Tytgat SH, and Lindeboom MY reviewed and revised the manuscript; Hulscher JB, Tytgat SH, and Lindeboom MY contributed to the writing; Weusten BL, Westerhof J, Brosens LA, Hulscher JB, Tytgat SH, and Lindeboom MY supervised the data collection and the progress the manuscript; Weusten BL, Westerhof J, and Brosens LA provided input to the study; Zwaveling S, Ruiterkamp J, Arets HG, Bittermann AJ, and van der Zee DC reviewed the study design, supervised the process and contributed to the interpretation of data; and all authors approved the final manuscript as submitted and agree to be accountable for all aspects of the work.
Institutional review board statement: This study was part of a larger cohort study on the long-term outcome in LGEA patients. The study protocol was submitted to the UMCU Ethics Committee (METC 18-458/C). According to the Medical Research Involving Human Subject Act, no ethical approval was required.
Informed consent statement: This study was part of a larger cohort study on the long-term outcome in LGEA patients. The study protocol was submitted to the UMCU Ethics Committee (METC 18-458/C). According to the Medical Research Involving Human Subject Act, no informed consent was required.
Conflict-of-interest statement: All the authors report no relevant conflicts of interest for this article.
Data sharing statement: No additional data are available.
STROBE statement: The authors have read the STROBE statement, and the manuscript was prepared and revised according to the STROBE statement.
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (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: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Eleonora Sofie van Tuyll van Serooskerken, MD, Researcher, Department of Pediatric Surgery, Wilhelmina Children’s Hospital, PO Box 85090, Utrecht 3508 AB, Netherlands. e.s.vantuyllvanserooskerken-3@umcutrecht.nl
Received: April 5, 2023
Peer-review started: April 5, 2023
First decision: June 1, 2023
Revised: June 15, 2023
Accepted: July 25, 2023
Article in press: July 25, 2023
Published online: September 16, 2023
ARTICLE HIGHLIGHTS
Research background

Previously, esophageal replacement (ER) with gastric pull-up (GPU) or jejunal interposition (JI) used to be the standard treatment for long-gap esophageal atresia (LGEA). Gastrointestinal symptoms are common in EA patients and may occur even more frequently after ER, due to a change of the anatomy.

Research motivation

Long-term macroscopic and microscopic graft changes are currently unknown and may be clinically relevant in patients with LGEA.

Research objectives

This study aims to evaluate clinical symptoms and macroscopic and microscopic graft changes in adolescence and adulthood.

Research methods

A cohort study including all LGEA patients ≥ 16 years who had undergone ER between 1985-2003 at two tertiary centers in the Netherlands was conducted. Clinical symptoms, contrast studies and endoscopies were collected prospectively.

Research results

Nine GPU patients and eleven JI patients were included in this study, with a median age of 21.5 years and 24.4 years respectively. Six of nine GPU patients (67%) reported reflux complaints and 64% of JI patients reported dysphagia symptoms. Dilatation of the jejunal graft was observed in 55%. Three GPU patients had columnar-lined epithelium and in two of these patients intestinal metaplasia was histologically confirmed.

Research conclusions

Long-term follow-up revealed significant macroscopic and microscopic graft changes after ER. Furthermore, this study revealed long-term clinical symptoms after both GPU and JI. GPU patients may have an increased risk on intestinal metaplasia. Dilatation of the graft and dysphagia symptoms were present in the majority of JI patients. Follow-up during adulthood after ER for LGEA is therefore suggested.

Research perspectives

This study highlights the importance of implementing an endoscopic follow-up program after ER for LGEA, particularly after GPU. Further investigations with larger patient cohorts are necessary to validate these findings.