Case Report
Copyright ©The Author(s) 2025. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Feb 26, 2025; 13(6): 98111
Published online Feb 26, 2025. doi: 10.12998/wjcc.v13.i6.98111
Saccharomyces boulardii as a single trigger of food protein-induced enterocolitis syndrome: Seven case reports
Jin-Bok Hwang, Hyo-Jeong Jang
Jin-Bok Hwang, Hyo-Jeong Jang, Department of Pediatrics, Keimyung University School of Medicine, Dongsan Medical Center, Daegu 42601, South Korea
Author contributions: Hwang JB was responsible for conceptualization, funding acquisition, and writing original draft; Hwang JB and Jang HJ were responsible for data curation, formal analysis, investigation, methodology, validation, visualization, writing review and editing; all authors have read and approved the final manuscript.
Supported by The Keimyung University Dongsan Medical Center in 2006.
Informed consent statement: This study was approved by the Keimyung University Dongsan Medical Center Institutional Review Board (No. 2022-05-103). Informed consent was waived because of the retrospective nature of the study.
Conflict-of-interest statement: Hwang JB reports that this work was supported by the research promoting grant from the Keimyung University Dongsan Medical Center in 2006. The other author has no conflict-of interest to declare.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
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: Hyo-Jeong Jang, MD, Associate Professor, Doctor, Department of Pediatrics, Keimyung University School of Medicine, Dongsan Medical Center, Dalgubeol-daero 1035, Dalseo-gu, Daegu 42601, South Korea. polarisjay@dsmc.or.kr
Received: June 18, 2024
Revised: September 12, 2024
Accepted: November 12, 2024
Published online: February 26, 2025
Processing time: 160 Days and 1.2 Hours
Abstract
BACKGROUND

Food protein-induced enterocolitis syndrome (FPIES) is the most serious type of non-immunoglobulin E (IgE)-mediated food allergic reaction manifesting as sepsis-like symptom, which can lead to shock. Saccharomyces boulardii (S. boulardii), a probiotic prescribed frequently in clinical settings, has been reported to trigger FPIES in an infant with soy-triggered FPIES. In this report, we describe a new clinical FPIES in which S. boulardii was the sole triggering factor of acute FPIES adverse reaction in seven healthy infants.

CASE SUMMARY

Seven FPIES cases triggered by only S. boulardii were gathered from 2011 to the present. None of the patients had previously experienced any allergic reaction to cow’s milk, soy, or complementary food. The age of the patients was 4-10-months old, and the symptoms of FPIES developed after ingestion of S. boulardii, which is mostly prescribed for the treatment of gastroenteritis or antibiotic-associated diarrhea. All patients experienced severe repetitive vomiting 1-3 hours after S. boulardii ingestion. Extreme lethargy, marked pallor, and cyanosis were also observed. No IgE-mediated hypersensitivity developed in any patient. Diarrhea was followed by initial intense vomiting in approximately 5-10 hours after S. boulardii ingestion, and only one case showed bloody, purulent, and foul-smelling diarrhea. The patients stabilized quickly, mostly within 6 hours. Symptoms got all improved within 24 hours after discontinuation of S. boulardii.

CONCLUSION

S. boulardii can be the sole trigger of acute FPIES and be prescribed cautiously even in healthy children without FPIES.

Keywords: Food protein-induced enterocolitis syndrome; Food hypersensitivity; Saccharomyces boulardii; Probiotics; Child; Case report

Core Tip: Saccharomyces boulardii (S. boulardii), one of the most commonly prescribed and generally safe probiotic for children, can trigger food protein-induced enterocolitis syndrome (FPIES) in patients with non-immunoglobulin E (IgE)-mediated or IgE-mediated food allergic reaction. Here, a new FPIES entity of healthy infants is described, in which acute FPIES is triggered solely by S. boulardii without any hypersensitivity to cow’s milk, soy, or complementary foods during follow-up. Those seven cases suggest that S. boulardii should be prescribed cautiously even in healthy children without FPIES to other foods.