Case Report
Copyright ©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Jan 16, 2023; 11(2): 456-463
Published online Jan 16, 2023. doi: 10.12998/wjcc.v11.i2.456
Systemic lupus erythematosus with multicentric reticulohistiocytosis: A case report
Ping-Ping Liu, Zong-Wen Shuai, Li Lian, Kang Wang
Ping-Ping Liu, Zong-Wen Shuai, Li Lian, Kang Wang, Department of Rheumatology and Immunology, Arthritis Research Institute, The First Affiliated Hospital of Anhui Medical University, Hefei 230000, Anhui Province, China
Author contributions: Liu PP and Wang K designed the study; Liu PP, Lian L, Shuai ZW and Wang K collected data and performed analyses; Liu PP drafted the manuscript; Wang K edited and revised manuscript; all authors contributed to the article and approved the submitted version.
Informed consent statement: Informed written consent was obtained from the patient.
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: Kang Wang, MD, Adjunct Professor, Doctor, Department of Rheumatology and Immunology, Arthritis Research Institute, The First Affiliated Hospital of Anhui Medical University, No. 218 Jixi Street, Hefei 230000, Anhui Province, China. wangkang057602@163.com
Received: October 17, 2022
Peer-review started: October 17, 2022
First decision: November 25, 2022
Revised: December 8, 2022
Accepted: December 23, 2022
Article in press: December 23, 2022
Published online: January 16, 2023
Processing time: 86 Days and 16.9 Hours
Abstract
BACKGROUND

Multicentric reticulohistiocytosis (MRH)/systemic lupus erythematosus (SLE) overlap syndrome is an uncommon disease in the clinic and is diagnosed through characteristic clinical manifestations, histopathology, and immunopathology. Here, we report the case of a 30-year-old woman with SLE who developed MRH.

CASE SUMMARY

A 30-year-old woman with a history of polyarthritis for the past 12 years had multiple skin nodules on her body for 10 years, including the sacrococcygeal area, dorsum of the hands, interphalangeal joint of the feet and sternoclavicular joint. The histopathology of a biopsy of the distal interphalangeal joint of the hands revealed granulomatous inflammation, fibrous hyperplasia with ground-glass degeneration, inflammatory cell exudation and focal necrosis. The immunohistochemical stains showed positive staining for CD68 and negative staining for S100 and acid-fast staining. The patient was diagnosed with SLE with MRH. Her symptoms were improved after a combined treatment of prednisone, hydroxychloroquine and cyclophosphamide.

CONCLUSION

MRH/SLE overlap syndrome is difficult to diagnose and treat. Cyclophosphamide may be an alternative choice for the treatment of MRH.

Keywords: Multicentric reticulohistiocytosis; Systemic lupus erythematosus; Cyclophosphamide; Systemic disorder; Case report

Core Tip: We present a case of systemic lupus erythematosus with multicentric reticulohistiocytosis that was reported for the second time. Multicentric reticulohistiocytosis/systemic lupus erythematosus overlap syndrome is an uncommon disease that is hard to diagnose and treat. This case illustrates how to diagnose and treat the comorbidities and the connection that exists between them. For treatment, cyclophosphamide may be an alternative choice for multicentric reticulohistiocytosis.