Case Report
Copyright ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Apr 6, 2020; 8(7): 1319-1325
Published online Apr 6, 2020. doi: 10.12998/wjcc.v8.i7.1319
Disseminated histoplasmosis in primary Sjögren syndrome: A case report
Jia-Ai Li, Ying-Ying Cheng, Zhi-Tao Cui, Wei Jiang, Wu-Qiong Zhang, Zhong-Hua Du, Bin Gao, Yin-Yin Xie, Hong-Mei Meng
Jia-Ai Li, Ying-Ying Cheng, Wei Jiang, Wu-Qiong Zhang, Bin Gao, Yin-Yin Xie, Hong-Mei Meng, Department of Neurology, First Hospital of Jilin University, Changchun 130021, Jilin Province, China
Zhi-Tao Cui, Department of Geriatrics, First Hospital of Jilin University, Changchun 130021, Jilin Province, China
Zhong-Hua Du, Department of Hematology, First Hospital of Jilin University, Changchun 130021, Jilin Province, China
Author contributions: Meng HM dominated this article; Li JA started and finished in writing and basic ideas; Cheng YY guided the completion of this article; Cui ZT provided a lot of guidance and corrections; Jiang W, Zhang WQ, Du ZH, Gao B, and Xie YY provided lots of help in the process of improving the article.
Supported by National Key R&D Program of China, No. 2017YFC0110304.
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: http://creativecommons.org/licenses/by-nc/4.0/
Corresponding author: Hong-Mei Meng, PhD, Doctor, Professor, Department of Neurology, First Hospital of Jilin University, 71 Xinmin Street, Changchun 130021, Jilin Province, China. hongmeiyp@126.com
Received: December 26, 2019
Peer-review started: December 26, 2019
First decision: February 26, 2020
Revised: March 24, 2020
Accepted: March 27, 2020
Article in press: March 27, 2020
Published online: April 6, 2020
Abstract
BACKGROUND

Sjögren syndrome (SS) is a chronic and systemic autoimmune disease characterized by lymphocytic infiltration of the exocrine glands. And histoplasmosis is an invasive mycosis caused by the saprophytic dimorphic fungus H. capsulatum. In patients with primary SS (PSS), disseminated histoplasmosis (DH) is extremely rare.

CASE SUMMARY

We report a 37-year-old female patient admitted to our hospital with exacerbating fatigue, somnolence, and pancytopenia as the main symptoms. She was eventually diagnosed with DH based on pancytopenia, splenomegaly, and findings of bone marrow smears. The atypical clinical symptoms made the diagnosis process more tortuous. Unfortunately, she died of respiratory failure on the day the diagnosis was confirmed.

CONCLUSION

We present a rare and interesting case of DH in a PSS patient. This case updates the geographic distribution of histoplasmosis in China, and expands the clinical manifestations of DH in PSS, highlighting the significance of constantly improving the understanding of PSS with DH.

Keywords: Sjögren syndrome, Infection, Disseminated histoplasmosis, Pancytopenia, Central nervous system involvement, Hematologic manifestations

Core tip: We present a rare and interesting case of disseminated histoplasmosis (DH) in a primary Sjögren syndrome (PSS) patient. This case updates the geographic distribution of histoplasmosis in China, and expands the clinical manifestations of DH in PSS. These findings improve our understanding of DH in PSS and may allow for earlier identification.