pISSN 2671-8790 eISSN 2671-8804

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Korean J Transplant

Published online November 9, 2021

© The Korean Society for Transplantation

Concurrent cytomegalovirus enteritis and atypical hemolytic uremic syndrome with gastrointestinal tract involvement: a case report

Min Hye Kim1 , Myung Hyun Cho2 , Yo Han Ahn1 , Jeong Mo Bae3 , Jin Soo Moon1 , Hee Gyung Kang1,4,5

1Department of Pediatrics, Seoul National University Children’s Hospital, Seoul, Korea
2Department of Pediatrics, Hallym University Sacred Heart Hospital, Anyang, Korea
3Department of Pathology, Seoul National University College of Medicine, Seoul, Korea
4Kidney Research Institute, Medical Research Center, Seoul National University College of Medicine, Seoul, Korea
5Wide River Institute of Immunology, Seoul National University, Hongcheon, Korea

Correspondence to: Hee Gyung Kang
Department of Pediatrics, Seoul National University Children’s Hospital, 103 Daehak-ro, Jongno-gu, Seoul 03080, Korea
Tel: +82-2-2072-0658
Fax: +82-2-743-3455
E-mail: kanghg@snu.ac.kr

Received: August 17, 2021; Revised: October 8, 2021; Accepted: October 8, 2021

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Atypical hemolytic uremic syndrome (aHUS) is a rare disease caused by complement dysregulation that may involve the extra-renal system. Without appropriate prophylactic treatment, aHUS commonly recur after kidney transplantation (KT). In contrast, cytomegalovirus (CMV) infection is common in KT recipients and may affect various organ systems. Herein, we report a case of recurrent aHUS complicated by CMV enteritis. This 17-year-old KT recipient with aHUS having a CFH mutation was admitted to the hospital for gastric pain and vomiting. With worsening hemogram, recurrence of aHUS involving the gastrointestinal (GI) system was suspected. Upon treatment with anti-C5 antibody, the patient’s blood counts soon improved, but her GI symptoms did not. Esophagogastroduodenoscopy revealed multiple ulcers in the duodenum with pathologic findings consistent with aHUS and CMV enteritis; however, she did not have CMV antigenemia despite these findings. Treatment with ganciclovir resolved GI symptoms within 7 days. This case shows that recurrence of aHUS is often induced by intercurrent infection, and common infections after allograft transplantation, such as CMV, should always be suspected and confirmed for a proper treatment, particularly because CMV enteritis may not accompany CMV antigenemia.

Keywords: Atypical hemolytic uremic syndrome, Cytomegalovirus, Kidney transplantation, Case report