Case Report
Copyright ©The Author(s) 2016. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. Dec 21, 2016; 22(47): 10465-10470
Published online Dec 21, 2016. doi: 10.3748/wjg.v22.i47.10465
Hepatosplenic T-cell lymphoma in a 47-year-old Crohn’s disease patient on thiopurine monotherapy
Maartje M van de Meeberg, Lauranne A A P Derikx, Harm A M Sinnige, Peet Nooijen, D Lucette Schipper, Loes H C Nissen
Maartje M van de Meeberg, D Lucette Schipper, Lauranne AAP Derikx, Loes HC Nissen, Department of Gastroenterology and Hepatology, Jeroen Bosch Hospital, 5200 ME ’s-Hertogenbosch, The Netherlands
Harm AM Sinnige, Department of Hematology, Jeroen Bosch Hospital, 5200 ME ’s-Hertogenbosch, The Netherlands
Peet Nooijen, Department of Pathology, Jeroen Bosch Hospital, 5200 ME ’s-Hertogenbosch, The Netherlands
Author contributions: van de Meeberg MM is first author and composed the manuscript; Derikx LAAP and Nissen LHC contributed equally in writing the manuscript; Sinnige HAM, Nooijen P and Schipper DL revised the manuscript; Schipper DL also provided the case.
Institutional review board statement: This case report was exempt.
Informed consent statement: The patient gave verbal informed consent prior to composing the manuscript.
Conflict-of-interest statement: All the authors have no conflicts of interests to declare.
Open-Access: 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:
Correspondence to: Maartje M van de Meeberg, MD, Department of Gastroenterology and Hepatology, Jeroen Bosch Hospital, Henri Dunantstraat 1, 5200 ME ’s-Hertogenbosch, The Netherlands.
Telephone: +31-73-5532000 Fax: +31-73-5538804
Received: June 26, 2016
Peer-review started: June 28, 2016
First decision: August 8, 2016
Revised: September 1, 2016
Accepted: September 28, 2016
Article in press: September 28, 2016
Published online: December 21, 2016

Hepatosplenic T-cell lymphoma (HSTCL) is a rare non-Hodgkin lymphoma with a high mortality rate. Higher incidence is reported in patients with inflammatory bowel disease, specifically in male patients that are younger than 35 years, and have been treated with thiopurine and tumor necrosis factor (TNF)-α inhibitor combination therapy for over 2 years. In this case report we describe a 47-year-old patient with Crohn’s disease (CD) who developed HSTCL after having been treated with thiopurine monotherapy for 14 years. To our best knowledge, only eleven cases exist of patients with CD who developed HSTCL while on thiopurine monotherapy. We report the first patient with CD, older than 35 years, who developed HSTCL while on thiopurine monotherapy. This emphasizes that HSTCL risk is not limited to young men receiving both thiopurines and TNF-α inhibitors.

Keywords: Hepatosplenic T-cell lymphoma, Thiopurine, Crohn’s disease, Immunosuppression

Core tip: In this manuscript we provide an overview of all known cases in literature with Crohn’s disease (CD) who developed hepatosplenic T-cell lymphoma (HSTCL) while on thiopurine monotherapy. In addition, we present our case of a patient with CD on thiopurine monotherapy who developed HSTCL at a relatively old age. This emphasizes that HSTCL may develop at all ages, even when the patient is solely on thiopurine monotherapy.