Case Report
Copyright ©The Author(s) 2025. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Hepatol. Apr 27, 2025; 17(4): 92426
Published online Apr 27, 2025. doi: 10.4254/wjh.v17.i4.92426
Hepatitis E virus infection-triggered intrahepatic cholestasis: A case report
Stephan Drexler, Frederic Haedge, Susanne N Weber, Marcin Krawczyk, Matthias S Matter, Carol I Geppert, Achim Weber, Bruno Stieger, Christian Trautwein, Andreas E Kremer
Stephan Drexler, Department of Medicine III, University Hospital Aachen, Aachen 52070, Germany
Frederic Haedge, Department of Medicine III, University Hospital RWTH Aachen, Aachen 52070, Germany
Susanne N Weber, Marcin Krawczyk, Department of Medicine II, Saarland University Medical Center, Saarland University, Homburg 66424, Germany
Marcin Krawczyk, Department of Gastroenterology, Hepatology and Transplant Medicine, Medical Faculty, University of Duisburg-Essen, Essen 45307, Germany
Matthias S Matter, Institute of Medical Genetics and Pathology, University Hospital Basel, University of Basel, Basel 4031, Switzerland
Carol I Geppert, Department of Pathology, Univ Erlangen Nurnberg, Erlangen D-91054, Germany
Carol I Geppert, Comprehensive Cancer Center Erlangen-EMN (CCC), University Hospital Erlangen, FAU Erlangen-Nuremberg, Erlangen D-91052, Germany
Achim Weber, Institute of Molecular Cancer Research, University Hospital Zurich and University Zurich, Zurich 8091, Switzerland
Bruno Stieger, Andreas E Kremer, Department of Gastroenterology and Hepatology, University Hospital Zürich, University of Zürich, Zurich 8091, Switzerland
Christian Trautwein, Department of Internal Medicine III, University Hospital RWTH Aachen, Aachen 52070, Germany
Andreas E Kremer, Department of Medicine 1, University Hospital Erlangen, Friedrich-Alexander-University Erlangen-Nürnberg, Erlangen 91054, Germany
Co-corresponding authors: Stephan Drexler and Andreas E Kremer.
Author contributions: Drexler S and Kremer AE contributed equally to this study as co-corresponding authors. Drexler S contributed to data collection and was involved in developing the research question and hypotheses; Haedge F was involved in data collection; Weber SN and Krawczyk M performed genotyping, next-generation sequencing, and interpretation of the results; Mater MS developed the research question and hypotheses; Geppert CI processed liver biopsy samples and performed tissue staining; Weber A also processed liver biopsy samples and performed various stainings; Stieger B performed the literature review and contributed to the development of the research question and hypotheses; Trautwein C was involved in data collection, and contributed to the development of the research question and hypotheses; Kremer AE was involved in data collection and development of the research question and hypotheses; all authors contributed to manuscript preparation as well as to the modifications during the revision.
Informed consent statement: Both patients provided informed consent in accordance with the ethical guidelines for the genetic analysis of the samples as well as for the publication of this case report.
Conflict-of-interest statement: Kremer AE has served as a consultant for AbbVie, Bayer, Beiersdorf, CymaBay Therapeutics, Escient, Dr. Falk, FMC, Gilead, GlaxoSmithKline, Guidepoint, Intercept Pharma, Medscape, Mirum, MSD, Myr, Roche, and Viofor, and received speaker’s honoraria from AbbVie, AOP Orphan, Bayer, Bristol Myers Squibb, CMS, CymaBay Therapeutics, Dr. Falk, Eisai, Eli Lilly, Gilead, GlaxoSmithKline, Intercept, Janssen, MSD, Newbridge, Novartis, and Zambon. M.S.M. has served as a consultant for ThermoFisher, Merck, GlaxoSmithKline, Janssen-Cilag, Roche, and Novartis and received speaker’s honoraria from Incyte Biosciences. Trautwein C: Grant support: DFG, BMBF, EU; Lectures: Falk, MSD, Sanofi, GSK, Ipsen; Advisory: Ipsen, MSD, GSK; Travel Support: Gilead, Abbvie. Apart from the declared conflicts of interest, the other authors assured that they do not have any disclosures to report.
CARE Checklist (2016) statement: We have read the CARE Checklist carefully, 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: Stephan Drexler, MD, Doctor, Department of Medicine III, University Hospital Aachen, Pauwelsstraße 30, Aachen 52070, Germany. sdrexler@ukaachen.de
Received: January 25, 2024
Revised: June 30, 2024
Accepted: July 9, 2024
Published online: April 27, 2025
Processing time: 455 Days and 18.9 Hours
Core Tip

Core Tip: This case report highlights the role of genetic variations in hepatobiliary transporters in predisposing individuals to intrahepatic cholestasis and identifies hepatitis E virus (HEV) as a potential trigger. We present two cases of HEV-triggered intrahepatic cholestasis in patients with polymorphisms in ATP8B1 and other transporter-related genes. These findings suggest that these genetic variants may act as predisposing factors for cholestatic episodes, underscoring the need for further research to confirm these results in larger patient cohorts and through functional studies.