Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Aug 6, 2022; 10(22): 7913-7923
Published online Aug 6, 2022. doi: 10.12998/wjcc.v10.i22.7913
Multicentric reticulohistiocytosis with prominent skin lesions and arthritis: A case report
Xiao-Li Xu, Xiao-Hong Liang, Juan Liu, Xu Deng, Lu Zhang, Zhi-Gang Wang
Xiao-Li Xu, Xiao-Hong Liang, Zhi-Gang Wang, Department of Geriatrics, The Third Affiliated Hospital of Soochow University, Changzhou 213003, Jiangsu Province, China
Juan Liu, Jiangsu Engineering Research Center for Tumor Immunotherapy, The Third Affiliated Hospital of Soochow University, Changzhou 213003, Jiangsu Province, China
Xu Deng, Department of Pathology, The Third Affiliated Hospital of Soochow University, Changzhou 213003, Jiangsu Province, China
Lu Zhang, Department of Rheumatology and Immunology, The Third Affiliated Hospital of Soochow University, Changzhou 213003, Jiangsu Province, China
Author contributions: Xu XL, Liang XH, and Liu J contributed to the conception of the study; Deng X participated in the pathological examination and analysis of the patients; Lu J performed the data analyses; Liang XH and Wang ZG helped perform the analysis with constructive discussions; Xu XL wrote the manuscript; all authors have read and approved the final manuscript.
Supported by the National Natural Science Foundation of China, No. 81770212; and Youth Science and Technology Project of Changzhou Health and Wellness Committee, No. QN201611.
Informed consent statement: Informed written consent was obtained from the patient for publication of this report and any accompanying images.
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 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: Zhi-Gang Wang, MD, Doctor, Department of Geriatrics, The Third Affiliated Hospital of Soochow University, No. 185 Juqian Street, Changzhou 213003, Jiangsu Province, China. wangzhigang_cz@163.com
Received: September 3, 2021
Peer-review started: September 3, 2021
First decision: December 1, 2021
Revised: December 14, 2021
Accepted: June 17, 2022
Article in press: June 17, 2022
Published online: August 6, 2022
Processing time: 321 Days and 23.7 Hours
Abstract
BACKGROUND

Multicentric reticulohistiocytosis (MRH) is a rare non-Langerhans histiocytosis of unknown etiology characterized by papulonodular skin lesions and progressive, erosive arthritis. To date, there have been approximately 300 cases of MRH reported worldwide. The majority of patients are Caucasian from western countries, and Asian patients are rare. Here, we report a case of MRH in a Chinese patient.

CASE SUMMARY

A 38-year-old male was admitted to the hospital with a rash that had persisted for over 2 years and bilateral knee pain for over 1 year. The patient’s symptoms had previously been misdiagnosed as eczema when there were only skin symptoms and was finally diagnosed as MRH after a skin biopsy of the left upper back. The patient was treated with glucocorticoids combined with an immunosuppressive regimen. While the skin lesions on both arms, abdomen, and back subsided, the skin lesions on the rest of the body did not increase. The interphalangeal joints of both thumbs and bilateral knee joints remained swollen and painful.

CONCLUSION

The case will help clinicians better identify and treat this disease in the absence of epidemiological studies or randomized controlled data.

Keywords: Multicentric reticulohistiocytosis; Papulonodular skin lesions; Arthritis; CD68; Case report

Core Tip: Multicentric reticulohistiocytosis (MRH) is a rare non-Langerhans histiocytosis of unknown etiology characterized by papulonodular skin lesions and progressive, erosive arthritis. To date, there have been approximately 300 cases of MRH reported worldwide. However, the majority of patients are Caucasian from western countries, and Asian patients are rare. Here, we report a case of MRH in a Chinese patient. We also review the relevant literature and comprehensively analyze the clinical characteristics of MRH. This case report and comprehensive analysis will help clinicians better identify and treat this disease in the absence of epidemiological studies or randomized controlled data.