Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Oct 16, 2022; 10(29): 10794-10802
Published online Oct 16, 2022. doi: 10.12998/wjcc.v10.i29.10794
Glucocorticoids combined with tofacitinib in the treatment of Castleman's disease: A case report
Xiao-Rui Liu, Mei Tian
Xiao-Rui Liu, Mei Tian, Department of Rheumatology and Immunology, Affiliated Hospital of Zunyi Medical University, Zunyi 563003, Guizhou Province, China
Author contributions: Liu XR wrote the manuscript; all authors have given final approval to the version to be submitted.
Informed consent statement: A written informed consent was obtained from the patient for publication of this case report.
Conflict-of-interest statement: The authors have nothing to disclose.
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: Mei Tian, PhD, Chief Doctor, Department of Rheumatology and Immunology, Affiliated Hospital of Zunyi Medical University, No. 149 Dalian Road, Huichuan District, Zunyi 563003, Guizhou Province, China. 348820517@qq.com
Received: July 7, 2022
Peer-review started: July 7, 2022
First decision: August 1, 2022
Revised: August 10, 2022
Accepted: September 5, 2022
Article in press: September 5, 2022
Published online: October 16, 2022
Abstract
BACKGROUND

Castleman's disease (CD), also known as vascular follicular lymphadenopathy is a rare proliferative disease of lymphoid tissue of unknown etiology that is clinically classified as unicentric CD (UCD) or multicentric CD (MCD) depending on lymph node involvement. At present, idiopathic MCD (iMCD) is treated with interleukin-6 inhibitors, but some patients have poor clinical outcomes. This paper reports on a case of iMCD that achieved a good therapeutic effect after treatment with glucocorticoids combined with tofacitinib. The relevant data are summarized and reported below.

CASE SUMMARY

This paper reports on a case of MCD in a 49-year-old female with persistent peritoneal effusion as the first manifestation and combined with multiple lymphadenopathies. Lymph node biopsy showed Castleman's disease-like changes. The ascites subsided after treatment with glucocorticoids and tofacitinib, indicating that the treatment was effective.

CONCLUSION

The combination of glucocorticoids with tofacitinib is an effective regimen for the treatment of CD.

Keywords: Castleman's disease, Multicentric Castleman's disease, Idiopathic multicentric Castleman's disease, Abdominal dropsy, Tofacitinib, Glucocorticoids, Case report

Core Tip: Castleman's disease (CD) is a rare proliferative disease of lymphoid tissue. The exact pathogenesis of CD remains unclear and is thought to be related to autoimmune diseases, viral infections and malignancies. The current preferred treatment for idiopathic multicentric CD (iMCD) is interleukin-6 inhibitors but there are still some patients who do not respond well to IL-6 inhibitors. In this paper, we report on a patient with iMCD having multiregional lymph node enlargement combined with malignant ascites who improved after treatment with janus kinase inhibitors.