Case Report
Copyright ©The Author(s) 2023. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Nov 16, 2023; 11(32): 7926-7934
Published online Nov 16, 2023. doi: 10.12998/wjcc.v11.i32.7926
Intracranial infection accompanied sweet’s syndrome in a patient with anti-interferon-γ autoantibodies: A case report
Jun-Hui Zheng, Dan Wu, Xiao-Yun Guo
Jun-Hui Zheng, Xiao-Yun Guo, General Internal Medicine, Affiliated Cancer Hospital of Guangxi Medical University, Nanning 530021, Guangxi Zhuang Autonomous Region, China
Dan Wu, Department of Intensive Care Unit, Affiliated Cancer Hospital of Guangxi Medical University, Nanning 530021, Guangxi Zhuang Autonomous Region, China
Co-first authors: Jun-Hui Zheng and Dan Wu.
Author contributions: Zheng JH and Wu D substantial contribution to the conception and design of the work; Zheng JH, Wu D and Guo XY contribution to the acquisition, analysis, interpretation of data for the work; Zheng JH contribution to article writing and revising; Guo XY agreement to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved; all authors have read and approve the final manuscript. Zheng JH and Wu D contributed equally to this work, they are substantial contribution to the conception and design of the work.
Supported by 2020 Guangxi University Young and Middle aged Teachers' Basic Research Ability Improvement Project, No. 2020KY03032.
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: Jun-Hui Zheng, Doctor, Associate Professor, General Internal Medicine, Affiliated Cancer Hospital of Guangxi Medical University, No. 71 Hedi Road, Nanning 530021, Guangxi Zhuang Autonomous Region, China. 13207811035@163.com
Received: September 26, 2023
Peer-review started: September 26, 2023
First decision: October 9, 2023
Revised: October 23, 2023
Accepted: November 9, 2023
Article in press: November 9, 2023
Published online: November 16, 2023
Processing time: 51 Days and 0.2 Hours
Abstract
BACKGROUND

Several reports of adult-onset immunodeficiency syndrome have been associated with anti-interferon-gamma (IFN-γ) autoantibodies (AIGAs). However, it is rare to find AIGAs with intracranial infections.

CASE SUMMARY

In this case study, we report a case of an AIGAs with intracranial infection and hand rashes considered Sweet’s syndrome. The patient presented to our hospital with a persistent cough, a fever that had been going on for 6 mo, and a rash that had been going on for a week. The patient started losing consciousness gradually on the fourth day after admission, with neck stiffness and weakened limb muscles. The upper lobe of the left lung had a high-density mass with no atypia and a few inflammatory cells in the interstitium. Brain magnetic resonance imaging and cerebrospinal fluid suggest intracranial infection. The pathology of the skin damage on the right upper extremity revealed an infectious lesion that was susceptible to Sweet’s disease. It has an anti-IFN-γ autoantibody titer of 1:2500. She was given empirical anti-non-tuberculous mycobacterial and antifungal treatments. The patient had no fever, obvious cough, headache, or rash on the hand. She got out of bed and took care of herself following hospitalization and discharge with medicine.

CONCLUSION

Adults with severe and recurrent infections of several organs should be considered for AIGAs if no other known risk factors exist. AIGAs are susceptible to subsequent intracranial infections and Sweet’s syndrome.

Keywords: Adult-onset immunodeficiency syndrome; Anti-interferon-gamma autoantibodies; Intracranial infection; Sweet disease; Case report

Core Tip: Anti-interferon-gamma (IFN-γ) autoantibodies (AIGAs) have been associated with adult-onset immunodeficiency syndrome. Most patients have multiple organ involvement upon presentation; lymph nodes are the most frequently affected organ, followed by the skin, lungs, bones, and joints. We describe a patient with AIGAs who also had an intracranial infection and hand rashes as Sweet’s syndrome. Anti-IFN-γ was increased despite the lack of a confirmed non-tuberculous mycobacterial (NTM) infection, and the empirical anti-NTM treatment in this patient was successful. Without recognized risk factors, AIGAs should be considered in patients with severe and recurrent infections of multiple organs. In AIGAs, subsequent intracranial infection and Sweet’s syndrome are possible.