Retrospective Cohort Study
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Apr 14, 2022; 28(14): 1455-1478
Published online Apr 14, 2022. doi: 10.3748/wjg.v28.i14.1455
Clinical classification of symptomatic heterotopic pancreas of the stomach and duodenum: A case series and systematic literature review
Michael T LeCompte, Brandon Mason, Keenan J Robbins, Motoyo Yano, Deyali Chatterjee, Ryan C Fields, Steven M Strasberg, William G Hawkins
Michael T LeCompte, Department of Surgical Oncology, University of North Carolina, Raleigh, NC 27608, United States
Brandon Mason, Department of Radiology, Stillwater Medical Center, Stillwater, OK 74074, United States
Keenan J Robbins, Department of General Surgery, Washington University St. Louis, St. Louis, MO 63110-8109, United States
Motoyo Yano, Department of Radiology, Mayo Clinic, Phoenix, AZ 8505, United States
Deyali Chatterjee, Department of Pathology and Immunology, MD Anderson Cancer Center, Houston, TX 77030, United States
Ryan C Fields, Department of Surgical Oncology, Washington University School of Medicine, St. Louis, MO 63110-8109, United States
Steven M Strasberg, William G Hawkins, Section of Hepatobiliary-Pancreatic and GI Surgery, Washington University St. Louis, St. Louis, MO 63110, United States
Author contributions: LeCompte M designed the study and performed the research, reviewed the manuscripts and wrote the paper; Mason B and Yano M reviewed the radiographic images and contributed content to the manuscript regarding radiographic identification; Chatterjee D collected and reviewed pathologic specimens; Robbins K assisted in reviewing medical records and collecting data; Hawkins G, Strasberg S, and Fields R participated in designing the study and contributed to the analysis and editing of the manuscript.
Institutional review board statement: This study was reviewed and approved by the institutional review board of Washington University of St. Louis. IRB number [#210811062].
Informed consent statement: Based on the study protocol and design, informed consent was determined to not be required and was therefore not obtained for this study. Patient information was collected and reviewed based on the parameters laid out by the IRB at our institution and was deidentified for analysis and reporting. The remainder of the study population was constructed based on a cohort of patients obtained through systematic review of the literature and was therefore based on patient information which had already been published and presented in the literature. For further questions or concerns please feel free to contact the corresponding author.
Conflict-of-interest statement: The authors of this study have no related conflicts of interest to disclose.
Data sharing statement: Technical data and information were provided by the corresponding author, Michael LeCompte who can be reached at michael_lecompte@med.unc.edu. Data from research subjects was anonymized and excluded any patient identifiers. Given the retrospective nature of this study, informed consent was deemed not necessary by the IRB. Additional data was collected and reviewed from datasets collected and published in the literature.
STROBE statement: The authors have read and revised the manuscript based on the STROBE guidelines.
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: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Michael T LeCompte, MD, Assistant Professor, Department of Surgical Oncology, University of North Carolina, 2800 Blue Ridge Rd. Suite 300, Raleigh, NC 27608, United States. mtleco01@gmail.com
Received: June 8, 2021
Peer-review started: June 8, 2021
First decision: June 27, 2021
Revised: July 11, 2021
Accepted: March 4, 2022
Article in press: March 4, 2022
Published online: April 14, 2022
Processing time: 302 Days and 6.4 Hours
ARTICLE HIGHLIGHTS
Research background

Heterotopic pancreas (HP) is a rare embryologic anomaly that occurs when aberrant pancreatic tissue develops independently from the main body of the main body of the pancreas. This can occur throughout the body and develop in various tissues however, this most commonly occurs in the stomach and duodenum. While the majority of these lesions are asymptomatic, many have the potential to cause harmful clinical manifestations.

Research motivation

Symptomatic heterotopic pancreas is difficult to identify and often mimics common upper gastrointestinal pathology making diagnosis and treatment challenging. Symptomatic cases of HP often require surgical or endoscopic management however, the optimal treatment strategy is poorly understood at this time and patients often undergo radical resection.

Research objectives

The purpose of this study was to review the cases of symptomatic HP at a single, high volume academic institution and compare these with a cohort of patients derived from a systematic review of symptomatic cases in the English literature. This combined cohort was used to classify the common presentation and clinical symptoms for HP and identify the various treatment options.

Research methods

A retrospective review was conducted of pathologic specimens containing heterotopic pancreas at a single, high volume, academic medical center. Symptomatic cases were identified and analyzed for details related to presentation, symptoms and treatment. A systematic review was then conducted utilizing Embase and PubMed databases for publications in English which pertained to heterotopic pancreas. Publications which included symptomatic cases of HP were included for final literature analysis and results were tabulated into a final cohort for review.

Research results

Institutional review: Twenty-nine cases of HP were identified at a single institution, of which, six presented with symptoms (20%). Two patients (7%) presented with elevated lipase and evidence of pancreatitis within the heterotopic lesion, two patients presented with gastrointestinal bleeding or anemia, one patient (3.4%) presented with perforated gastric ulceration related to a heterotopic lesion, and one patient presented with gastric outlet obstruction from a HP lesion located at the pylorus.

Systematic review: Literature review resulted in identification of 1220 unique publications related to HP of which 232 publications met inclusion criteria and were reviewed. The final cohort of patients was 934 patients. Analysis of symptomatic cases revealed that the majority of cases could be classified into the following: (1) Dyspepsia, which included symptoms of epigastric pain, eructation, nausea or bloating with meals (n = 445, 48%); (2) Pancreatitis within the ectopic lesion (n = 260, 28%); (3) Gastrointestinal bleeding (n = 80, 9%); and (4) Gastric outlet obstruction (n = 80, 9%). 37 patients presented with jaundice and biliary obstruction due to periampullary lesions (4%), and 3 patients presented with perforated ulcers. The majority of patients underwent surgical or endoscopic resection (n = 832, 90%) and 85% of reported cases indicated symptom resolution. However, most patients underwent some form of formal anatomic resection (n = 490, 61%) most commonly related to improper diagnosis preoperatively and few benefited from local surgical or endoscopic resection.

Research conclusions

Heterotopic pancreas is a rare condition but has the ability to manifest severe clinical symptoms. These most commonly arise in the stomach and duodenum and can be classified within for main categories. Improved recognition and diagnosis of this disease process may help guide treatment recommendations and improve outcomes for patients.

Research perspectives

Prospective evaluation of medical and interventional therapies is needed to determine the most appropriate treatment for the different manifestations for heterotopic pancreas.