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
Abstract
BACKGROUND

Esophageal replacement (ER) with gastric pull-up (GPU) or jejunal interposition (JI) used to be the standard treatment for long-gap esophageal atresia (LGEA). Changes of the ER grafts on a macro- and microscopic level however, are unknown.

AIM

To evaluate long-term clinical symptoms and anatomical and mucosal changes in adolescents and adults after ER for LGEA.

METHODS

A cohort study was conducted including all LGEA patients ≥ 16 years who had undergone GPU or JI between 1985-2003 at two tertiary referral centers in the Netherlands. Patients underwent clinical assessment, contrast study and endoscopy with biopsy. Data was collected prospectively. Group differences between JI and GPU patients, and associations between different outcome measures were assessed using the Fisher’s exact test for bivariate variables and the Mann-Whitney U-test for continuous variables. Differences with a P-value < 0.05 were considered statistically significant.

RESULTS

Nine GPU patients and eleven JI patients were included. Median age at follow-up was 21.5 years and 24.4 years, respectively. Reflux was reported in six GPU patients (67%) vs four JI patients (36%) (P = 0.37). Dysphagia symptoms were reported in 64% of JI patients, compared to 22% of GPU patients (P = 0.09). Contrast studies showed dilatation of the jejunal graft in six patients (55%) and graft lengthening in four of these six patients. Endoscopy revealed columnar-lined esophagus in three GPU patients (33%) and intestinal metaplasia was histologically confirmed in two patients (22%). No association was found between reflux symptoms and macroscopic anomalies or intestinal metaplasia. Three GPU patients (33%) experienced severe feeding problems vs none in the JI group. The median body mass index of JI patients was 20.9 kg/m2vs 19.5 kg/m2 in GPU patients (P = 0.08).

CONCLUSION

The majority of GPU patients had reflux and intestinal metaplasia in 22%. The majority of JI patients had dysphagia and a dilated graft. Follow-up after ER for LGEA is essential.

Keywords: Long-gap esophageal atresia, Jejunal interposition, Gastric pull-up, Barrett’s esophagus, Intestinal metaplasia, Esophageal replacement

Core Tip: Long-gap esophageal atresia (LGEA) remains a surgical challenge. Preservation of the native esophagus in LGEA is the treatment of choice. Previously however, almost all LGEA patients underwent esophageal replacement (ER). This study evaluated long-term clinical symptoms and anatomical and mucosal changes in adolescents and adults after ER for LGEA. We found that long-term symptoms and graft alterations were common. The majority of gastric pull-up patients had reflux symptoms with intestinal metaplasia in 22%. The majority of jejunal interposition (JI) patients had dysphagia symptoms and more than half of the JI grafts were dilated.