Case Report
Copyright ©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jul 6, 2021; 9(19): 5270-5279
Published online Jul 6, 2021. doi: 10.12998/wjcc.v9.i19.5270
Maintenance treatment with infliximab for ulcerative ileitis after intestinal transplantation: A case report
Takumi Fujimura, Yohei Yamada, Tomoshige Umeyama, Yumi Kudo, Hiroki Kanamori, Teizaburo Mori, Takahiro Shimizu, Mototoshi Kato, Miho Kawaida, Naoki Hosoe, Yasushi Hasegawa, Kentaro Matsubara, Naoki Shimojima, Masahiro Shinoda, Hideaki Obara, Makoto Naganuma, Yuko Kitagawa, Ken Hoshino, Tatsuo Kuroda
Takumi Fujimura, Department of Pediatric Surgery, National Saitama Hospital, Wako Shi, Saitama 351-0102, Japan
Takumi Fujimura, Yohei Yamada, Tomoshige Umeyama, Yumi Kudo, Hiroki Kanamori, Teizaburo Mori, Takahiro Shimizu, Mototoshi Kato, Naoki Shimojima, Ken Hoshino, Tatsuo Kuroda, Department of Pediatric Surgery, Keio University School of Medicine, Tokyo 160-8582, Japan
Miho Kawaida, Department of Pathology, Keio University School of Medicine, Tokyo 160-8582, Japan
Naoki Hosoe, Center for Diagnostic and Therapeutic Endoscopy, Keio University School of Medicine, Tokyo 160-8582, Japan
Yasushi Hasegawa, Kentaro Matsubara, Masahiro Shinoda, Hideaki Obara, Yuko Kitagawa, Department of Surgery, Keio University School of Medicine, Tokyo 160-8582, Japan
Masahiro Shinoda, Digestive Diseases Center, International University of Health and Welfare, Mita Hospital, Tokyo 108-8329, Japan
Makoto Naganuma, Department of Gastroenterology and Hepatology, Keio University School of Medicine, Tokyo 160-8582, Japan
Author contributions: Fujimura T wrote and coordinated this manuscript; Yamada Y, Kuroda T, Kitagawa Y, Hoshino K, Hasegawa Y, Matsubara K, Shimojima N, Shinoda M, Obara H, and Naganuma M helped draft the manuscript; Umeyama T, Kudo Y, Kanamori H, Mori T, Shimizu T, Kato M, and Hosoe N helped treatment of the patient and corrected data; Kawaida M performed the pathological analysis; all authors read and approved the final manuscript.
Supported by JSPS KAKENHI, No. JP18K16286, and No. JP18K08600.
Informed consent statement: Consent was obtained from the patient for the publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflicts of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), 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: http://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Yohei Yamada, FACS, MD, PhD, Assistant Lecturer, Department of Pediatric Surgery, Keio University School of Medicine, 35 Shinanomachi, Shinjuku-ku, Tokyo 160-8582, Japan. yohei.z7@keio.jp
Received: February 3, 2021
Peer-review started: February 3, 2021
First decision: March 6, 2021
Revised: March 18, 2021
Accepted: May 17, 2021
Article in press: May 17, 2021
Published online: July 6, 2021
Processing time: 141 Days and 4.3 Hours
Abstract
BACKGROUND

Evidence has been published on the successful applications of the anti-tumor necrosis factor alpha antibody infliximab, such as induction therapy, salvage treatment for acute cellular rejection, and treatment for chronic ulcerative inflammation, in intestinal transplant recipients. However, the optimal protocol for the effective use of infliximab remains largely undetermined due to scarcity of available clinical data. We report a continuative application of infliximab as maintenance therapy for recurrent chronic ulcerative ileitis in a recipient of isolated intestinal transplantation (ITx).

CASE SUMMARY

The patient was a 11-year-old boy with intestinal motility disorder classified as a hypogenic type of intestinal dysganglionosis. The patient underwent living-donor related intestinal transplant. His immunosuppression regimen consisted of daclizumab, tacrolimus, and steroids. Although he did not show rejection while on tacrolimus monotherapy, routine screening endoscopy showed several ulcerative lesions in the distal end of the graft 2 years after the intestinal transplant. Endoscopic work up to evaluate the progression of anemia revealed stenosis with ulcerative inflammatory changes and multiple longitudinal ulcers in the graft. Since the endoscopic findings suggested ulcerative lesions in Crohn’s disease, infliximab treatment was considered. Treatment with infliximab and a small dose of oral prednisolone afforded successful withdrawal of total parenteral nutrition and maintenance of a well-functioning graft without infectious complications for 5 years since the administration of the first dose of infliximab.

CONCLUSION

Infliximab is effective as maintenance therapy for recurrent chronic ulcerative ileitis in an isolated ITx patient.

Keywords: Intestinal transplantation; Chronic ulcer; Infliximab; Crohn’s disease; Tumor necrosis factor alpha; Case report

Core Tip: Infliximab binds to soluble and transmembrane forms of human tumor necrosis factor alpha (TNF-α). Ulcerative inflammatory changes in the graft under intestinal transplantation (ITx) is an often-encountered finding. However, it does not meet the criteria for so-called rejection and is close to the pathology of Crohn’s disease. Studies in Crohn’s disease patients revealed that anti-TNF-α therapy provides better outcomes when combined with immunomodulatory agents and that therapeutic drug monitoring might help optimize dosing. Infliximab may be effective as a treatment for ulcerative inflammation in the intestinal graft that does not meet the criteria for acute cellular rejection not improved by immunosuppressant conditioning. The optimal management for recurrent ulcerative inflammation under ITx settings by using anti-TNF-α therapy needs further elucidation.