Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Sep 6, 2022; 10(25): 9004-9011
Published online Sep 6, 2022. doi: 10.12998/wjcc.v10.i25.9004
Coexistent Kaposi sarcoma and post-transplant lymphoproliferative disorder in the same lymph nodes after pediatric liver transplantation: A case report
Shu-Hong Zhang, Guang-Yong Chen, Zhi-Jun Zhu, Lin Wei, Ying Liu, Jing-Yi Liu
Shu-Hong Zhang, Guang-Yong Chen, Department of Pathology, Beijing Friendship Hospital, Capital Medical University, Beijing 100050, China
Zhi-Jun Zhu, Lin Wei, Ying Liu, Jing-Yi Liu, Liver Transplantation Center, National Clinical Research Center for Digestive Diseases, Beijing Friendship Hospital, Capital Medical University, Beijing 100050, China
Zhi-Jun Zhu, Lin Wei, Ying Liu, Jing-Yi Liu, Clinical Center for Pediatric Liver Transplantation, Capital Medical University, Beijing 100050, China
Ying Liu, Jing-Yi Liu, Department of Critical Liver Diseases, Liver Research Center, Beijing Friendship Hospital, Capital Medical University, Beijing 100050, China
Author contributions: Zhang SH reviewed the literature, drafted the manuscript, and revised the manuscript for important intellectual content; Cheng GY collected the pathological data; Zhu ZJ, Wei L, Liu Y, and Liu JY provided and analyzed the clinical data; all authors have read and approve the final manuscript.
Supported by the Digestive Medical Coordinated Development Center of Beijing Hospitals Authority, No. XXX0102.
Informed consent statement: Informed written consent was obtained from the patient’s parents for publication of this report and any accompanying images.
Conflict-of-interest statement: All authors declare that they have no conflict 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: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Shu-Hong Zhang, MD, Doctor, Department of Pathology, Beijing Friendship Hospital, Capital Medical University, No. 95 Yong-an Road, Xicheng District, Beijing 100050, China. 13552880233@163.com
Received: March 14, 2022
Peer-review started: March 14, 2022
First decision: April 8, 2022
Revised: April 24, 2022
Accepted: July 29, 2022
Article in press: July 29, 2022
Published online: September 6, 2022
Processing time: 165 Days and 5.1 Hours
Abstract
BACKGROUND

Kaposi sarcoma and post-transplant lymphoproliferative disorder have been occasionally reported in post-liver transplant patients. However, the simultaneous occurrence of these two diseases in the same lymph nodes is very rare.

CASE SUMMARY

We report the case of a 19-mo-old boy, who presented with intermittent fever and enlarged cervical lymph nodes after liver transplantation. Six cervical lymph nodes were biopsied, and the histopathological examinations revealed multifocal hyperplasia of spindle cells around small blood vessels, extravasated erythrocytes, and heavy infiltration of plasma cells in the cortex and medulla of the lymph nodes. The immunohistochemical analyses of spindle cells revealed positive expression of CD34, CD31, erythroblast transformation-specific-related gene, friend leukemia integration 1, and human herpesvirus-8. The lymphoproliferative lesions expressed CD38, CD138, and multiple myeloma 1. Epstein-Barr encoded RNA in situ hybridization demonstrated Epstein-Barr virus-positive lymphoid cells. Finally, we diagnosed the coexistence of Kaposi sarcoma and post-transplant lymphoproliferative disorder (plasmacytic hyperplasia) in the same lymph nodes. Treatment strategy included anti-CD20 monoclonal antibody (rituximab) and discontinuation of the immunosuppressant therapies. Lymph node biopsies during follow-up examinations revealed lymphoid hyperplasia.

CONCLUSION

The rare coexistence of Kaposi sarcoma and post-transplant lymphoproliferative disorder in the same lymph nodes post-liver transplantation possibly associates with immunodeficiency and Epstein-Barr virus and human herpesvirus-8 coinfection.

Keywords: Kaposi sarcoma, Post-transplant lymphoproliferative disorder, Liver transplantation, Epstein-Barr virus infections, Human herpesvirus-8, Case report

Core Tip: We report a rare case of coexistent Kaposi sarcoma and post-transplant lymphoproliferative disorder in the same lymph nodes after a pediatric liver transplant. The definitive diagnosis was based on histopathological examination of the lymph nodes. The patient recovered after discontinuation of the immunosuppressants and application of anti-CD20 monoclonal antibody (rituximab) therapy. In conclusion, the concurrent occurrence of these two disorders may be associated with immunodeficiency as well as Epstein-Barr virus and human herpesvirus-8 coinfection.