Schult A, Andersson M, Asin-Cayuela J, Olsson KS. Hemorrhagic colitis induced by trientine in a 51-year-old patient with Wilson’s disease waiting for liver transplantation: A case report. World J Hepatol 2022; 14(8): 1687-1691 [PMID: 36157863 DOI: 10.4254/wjh.v14.i8.1687]
Corresponding Author of This Article
Andreas Schult, MD, Consultant Physician-Scientist, Transplant Institute, Sahlgrenska University Hospital, Bruna Stråket 5, Gothenburg 41345, Sweden. andreas.schult@vgregion.se
Research Domain of This Article
Gastroenterology & Hepatology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (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/
World J Hepatol. Aug 27, 2022; 14(8): 1687-1691 Published online Aug 27, 2022. doi: 10.4254/wjh.v14.i8.1687
Hemorrhagic colitis induced by trientine in a 51-year-old patient with Wilson’s disease waiting for liver transplantation: A case report
Andreas Schult, Matts Andersson, Jorge Asin-Cayuela, Karl Sigvard Olsson
Andreas Schult, Transplant Institute, Sahlgrenska University Hospital, Gothenburg 41345, Sweden
Andreas Schult, Department of Molecular and Clinical Medicine, Institute of Medicine, Sahlgrenska Academy, University of Gothenburg, Gothenburg 41345, Sweden
Matts Andersson, Department of Medicine, Örnsköldsvik Hospital, Örnsköldsvik 89145, Sweden
Jorge Asin-Cayuela, Department of Clinical Genetics, Sahlgrenska University Hospital, Gothenburg 41345, Sweden
Karl Sigvard Olsson, Section of Hematology and Coagulation, Department of Medicine and Clinical Nutrition, Institute of Medicine, Sahlgrenska Academy, University of Gothenburg, Gothenburg 41345, Sweden
Author contributions: Schult A wrote the part of the manuscript concerning transplantation, pretransplant work-up and revised the manuscript; Andersson M contributed to pretransplant clinical information; Asin-Cayuela J wrote the part concerning genetic analyses and critically revised the manuscript; Olsson KS drafted the main manuscript and critically revised the manuscript.
Informed consent statement: Written informed consent was obtained from the patient for publication of this case report.
Conflict-of-interest statement: All authors have nothing to declare.
CARE Checklist (2016) statement: The current case report follows 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: Andreas Schult, MD, Consultant Physician-Scientist, Transplant Institute, Sahlgrenska University Hospital, Bruna Stråket 5, Gothenburg 41345, Sweden. andreas.schult@vgregion.se
Received: March 3, 2022 Peer-review started: March 3, 2022 First decision: June 8, 2022 Revised: July 1, 2022 Accepted: July 27, 2022 Article in press: July 27, 2022 Published online: August 27, 2022 Processing time: 175 Days and 18.4 Hours
Abstract
BACKGROUND
Wilson’s disease (WD) is a rare inherited disorder of copper metabolism. Treatment consists of chelating agents, but side effects are common. We describe a patient who developed colitis during trientine treatment leading to decompensation of liver cirrhosis.
CASE SUMMARY
A healthy 51-year-old woman was diagnosed with liver cirrhosis due to decompensation with ascites. Etiologic evaluation raised suspicion of hereditary hemochromatosis because of compound heterozygosity HFE p.C282Y/p.H63D, and phlebotomy was started. Re-evaluation showed low ceruloplasmin, increased urinary copper excretion and the presence of Kayser-Fleischer rings. WD was confirmed by genetic analysis. Because of decompensated cirrhosis, she was referred for liver transplant evaluation. Simultaneously, treatment with trientine was initiated. Liver function initially stabilized, and the patient was not accepted for a liver transplant. Shortly after this, she developed severe hemorrhagic colitis, most probably a side effect of trientine. During that episode, she decompensated with hepatic encephalopathy. Because of a second decompensating event, she was accepted for liver transplantation, and an uneventful transplantation was carried out after clinical improvement of colitis.
CONCLUSION
Despite WD being a rare disorder, it is important to consider because it can present with a plethora of symptoms from childhood to an elderly age. Colitis should be recognized as a serious adverse drug reaction to trientine treatment that can result in decompensated liver disease.
Core Tip: Even if Wilson’s disease is a rare disorder, it is important to consider as a cause of liver disease. Treatment with chelating agents is associated with multiple side effects, and colitis should be recognized as a serious adverse drug reaction to trientine. Such a serious adverse event can trigger hepatic decompensation with the need for liver transplantation.
