Miyaguchi K, Yamaoka M, Tsuzuki Y, Ashitani K, Ohgo H, Miyagawa Y, Ishizawa K, Kayano H, Nakamoto H, Imaeda H. Epstein–Barr virus-associated hemophagocytic syndrome in a patient with ulcerative colitis during treatment with azathioprine: A case report and review of literature. World J Clin Cases 2018; 6(14): 776-780 [PMID: 30510942 DOI: 10.12998/wjcc.v6.i14.776]
Corresponding Author of This Article
Yoshikazu Tsuzuki, FACG, MD, PhD, Associate Professor, Department of Gastroenterology, Saitama Medical University, 38 Morohongo, Moroyama-machi, Iruma-gun 350-0495, Saitama, Japan. ytsuzuki@saitama-med.ac.jp
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 Clin Cases. Nov 26, 2018; 6(14): 776-780 Published online Nov 26, 2018. doi: 10.12998/wjcc.v6.i14.776
Epstein–Barr virus-associated hemophagocytic syndrome in a patient with ulcerative colitis during treatment with azathioprine: A case report and review of literature
Kazuya Miyaguchi, Minoru Yamaoka, Yoshikazu Tsuzuki, Keigo Ashitani, Hideki Ohgo, Yoshitaka Miyagawa, Hidetomo Nakamoto, Hiroyuki Imaeda, Department of General Internal Medicine, Saitama Medical University, Iruma-gun 350-0495, Saitama, Japan
Yoshikazu Tsuzuki, Hideki Ohgo, Hiroyuki Imaeda, Department of Gastroenterology, Saitama Medical University, Iruma-gun 350-0495, Saitama, Japan
Keisuke Ishizawa, Hidekazu Kayano, Department of Pathology, Saitama Medical University, Iruma-gun 350-0495, Saitama, Japan
Author contributions: Miyaguchi K was involved in data collection, data analysis, and writing up the first draft of the paper; Tsuzuki Y contributed towards data analysis, and patient follow-up in the out-patient unit; Yamaoka M contributed to medical treatment; Imaeda H supervised, and finalized the manuscript; all authors approved the final manuscript.
Informed consent statement: Written informed consent was obtained from the patient.
Conflict-of-interest statement: The authors have no conflicts of interest 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: http://creativecommons.org/licenses/by-nc/4.0/
Correspondence to: Yoshikazu Tsuzuki, FACG, MD, PhD, Associate Professor, Department of Gastroenterology, Saitama Medical University, 38 Morohongo, Moroyama-machi, Iruma-gun 350-0495, Saitama, Japan. ytsuzuki@saitama-med.ac.jp
Telephone: +81-49-2761667 Fax: +81-49-2761667
Received: June 22, 2018 Peer-review started: June 22, 2018 First decision: August 1, 2018 Revised: August 18, 2018 Accepted: October 8, 2018 Article in press: October 8, 2018 Published online: November 26, 2018 Processing time: 157 Days and 14.8 Hours
Abstract
A 19-year-old female was diagnosed with ulcerative colitis when she presented with persistent melena, and has been treated with 5-aminosalicylic acid for 4 years, with additional azathioprine for 2 years at our hospital. The patient experienced high-grade fevers, chills, and cough five d prior to presenting to the outpatient unit. At first, the patient was suspected to have developed neutropenic fever; however, she was diagnosed with Epstein-Barr virus-associated hemophagocytic syndrome (EB-VAHS) upon fulfilling the diagnostic criteria after bone marrow aspiration. When patients with inflammatory bowel disease treated with immunomodulators, such as thiopurine preparations, develop fever, EB-VAHS should be considered in the differential diagnosis.
Core tip: A 19-year-old female was diagnosed with ulcerative colitis, and has been receiving treatment with 5-aminosalicylic acid for 4 years, with additional azathioprine for 2 years. The patient experienced high-grade fever five d prior to presenting to the outpatient unit. She was diagnosed with Epstein-Barr virus-associated hemophagocytic syndrome (VAHS). VAHS is one of the rare and life-threatening pathophysiological conditions induced by thiopurine treatment. In such cases, early diagnosis is necessary, along with management of therapy, and related complications. When patients with inflammatory bowel disease, treated with immunomodulators such as thiopurine preparations, demonstrate high fever, VAHS should be considered in the differential diagnosis.
