Case Report
Copyright ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Nov 26, 2020; 8(22): 5663-5669
Published online Nov 26, 2020. doi: 10.12998/wjcc.v8.i22.5663
Oral granuloma in a pediatric patient with chronic graft-versus-host disease: A case report
Atsushi Uesugi, Fumihiko Tsushima, Makoto Kodama, Takeshi Kuroshima, Jinkyo Sakurai, Hiroyuki Harada
Atsushi Uesugi, Fumihiko Tsushima, Takeshi Kuroshima, Jinkyo Sakurai, Hiroyuki Harada, Oral and Maxillofacial Surgery, Tokyo Medical and Dental University, Tokyo 113-8549, Japan
Makoto Kodama, Human Pathology, Tokyo Medical and Dental University, Tokyo 113-8510, Japan
Author contributions: Atsushi U, Kuroshima T, and Tsushima F were the patient’s oral and maxillofacial surgeons, reviewed the literature, and contributed to manuscript drafting; Sakurai J and Harada H were the patient’s oral and maxillofacial surgeons, reviewed the literature, and drafted the manuscript; Kodama M performed pathological analysis, reviewed the literature, and drafted the manuscript. All authors issued final approval for the version to be submitted.
Informed consent statement: The authors got informed consent from the patient's family.
Conflict-of-interest statement: The authors declare that they have no conflict of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016) and have prepared and revised the manuscript 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: Atsushi Uesugi, DDS, PhD, Medical Assistant, Oral and Maxillofacial Surgery, Tokyo Medical and Dental University, 1-5-45 Yushima, Bunkyo-ku, Tokyo 113-8549, Japan. uesugi.atsushi@tokushima-u.ac.jp
Received: June 9, 2020
Peer-review started: June 9, 2020
First decision: September 24, 2020
Revised: October 4, 2020
Accepted: October 26, 2020
Article in press: October 26, 2020
Published online: November 26, 2020
Processing time: 161 Days and 8.4 Hours
Abstract
BACKGROUND

Oral mucositis is often observed with graft-versus-host disease (GVHD); however, the occurrence of oral granuloma is rare. The rapid increase in granulomatous lesions should be distinguished from malignant tumors in patients with GVHD because malignant diseases can develop in those patients. This case is the youngest pediatric patient with granuloma associated with GVHD.

CASE SUMMARY

The patient was a 1-year and 5-mo-old girl who presented to our department for the management of oral nodules. At the age of 5 mo, she was diagnosed with primary immunodeficiency disease, cord blood transplant was performed at 11 mo and bone marrow transplant at 1 year of age. After transplantation, GVHD and oral mucositis developed, and tacrolimus was administered. Interestingly, nodules appeared on the lower lip and buccal mucosa, which spontaneously disappeared. Then, a new nodule appeared on the left lateral border of the tongue. Resection was performed and the histopathological diagnosis was granuloma. The origin of these nodules were considered to be the fibroblasts activated under inflammation caused by GVHD because the calcineurin inhibitor tacrolimus acted on their proliferation.

CONCLUSION

It is very important to distinguish oral granulomatous lesions from malignancies if GVHD is present at the base and if immunosuppressive agents and steroids are being administered.

Keywords: Oral granuloma; Graft-versus-host disease; Bone marrow transplantation; Primary immunodeficiency disease; Tacrolimus; Case report

Core Tip: Oral mucositis is often observed with graft-versus-host disease (GVHD); however, the presence of oral granuloma is rare. This case is the youngest pediatric patient with granuloma associated with GVHD. At the age of 5 mo, she was diagnosed with primary immunodeficiency disease. After transplantation, she developed GVHD and oral mucositis. Notably, nodules appeared on the lip and buccal mucosa, which spontaneously disappeared. Then, a new nodule appeared on the tongue. Histopathological diagnosis was granuloma. It is important to distinguish rapidly grown granulomatous nodules from malignant tumors.