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Open Access 01-12-2024 | Pathology | Case Report

Multiple pathogen infections accompanied with ocular pathologies in a patient with high-titer Anti-IFN-γ autoantibodies: a case report

Authors: Siyao Wu, Siqiao Liang, Hanlin Liang, Yan Ning, Xia Li, Zhiyi He

Published in: BMC Infectious Diseases | Issue 1/2024

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Abstract

The typical clinical characteristic of patients with anti-IFN-γ autoantibodies (AIGAs) is primarily associated with infection caused by intracellular pathogens. With continued research, additional clinical characteristics have been gradually uncovered. Here, we present a case of multiple pathogen infections accompanied by ocular pathologies in a patient with high titers of AIGAs. The patient, a 53-year-old female patient, was admitted to our hospital after finding a mass in the right supraclavicular fossa. She was successively diagnosed with Talaromyces marneffei, Aspergillus flavus and Nontuberculous mycobacteria (NTM) infections. Then, she received a complete course of antifungal agents for nearly 3.5 years and anti-NTM treatment for nearly 3 years, with discontinuation upon symptom improvement. However, there was a rapid recurrence of the infection upon cessation of the drug despite improvement in the patient’s symptoms. Moreover, when the recurrent infection stabilized, the patient exhibited immune conjunctivitis and dry eye, which was successfully treated by tacrolimus eye drops and lubricant. Patients with high-titer AIGAs are more prone to experiencing recurrence and/or persistent infection, as well as immune disorders.
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Literature
1.
go back to reference Browne SK, Burbelo PD, Chetchotisakd P, Suputtamongkol Y, Kiertiburanakul S, Shaw PA, et al. Adult-onset immunodeficiency in Thailand and Taiwan. N Engl J Med. 2012;367(8):725–34.CrossRefPubMedPubMedCentral Browne SK, Burbelo PD, Chetchotisakd P, Suputtamongkol Y, Kiertiburanakul S, Shaw PA, et al. Adult-onset immunodeficiency in Thailand and Taiwan. N Engl J Med. 2012;367(8):725–34.CrossRefPubMedPubMedCentral
2.
go back to reference Guo J, Ning XQ, Ding JY, Zheng YQ, Shi NN, Wu FY, et al. Anti-IFN-γ autoantibodies underlie disseminated Talaromyces marneffei infections. J Exp Med. 2020;217(12):e20190502.CrossRefPubMedPubMedCentral Guo J, Ning XQ, Ding JY, Zheng YQ, Shi NN, Wu FY, et al. Anti-IFN-γ autoantibodies underlie disseminated Talaromyces marneffei infections. J Exp Med. 2020;217(12):e20190502.CrossRefPubMedPubMedCentral
3.
go back to reference Qiu Y, Fang G, Ye F, Zeng W, Tang M, Wei X, et al. Pathogen spectrum and immunotherapy in patients with anti-IFN-γ autoantibodies: a multicenter retrospective study and systematic review. Front Immunol. 2022;13:1051673.CrossRefPubMedPubMedCentral Qiu Y, Fang G, Ye F, Zeng W, Tang M, Wei X, et al. Pathogen spectrum and immunotherapy in patients with anti-IFN-γ autoantibodies: a multicenter retrospective study and systematic review. Front Immunol. 2022;13:1051673.CrossRefPubMedPubMedCentral
4.
go back to reference Liang X, Si L, Li Y, Zhang J, Deng J, Bai J, et al. Talaromyces marneffei infection relapse presenting as osteolytic destruction followed by suspected nontuberculous mycobacterium infection during 6 years of follow-up: a case update. Int J Infect Dis. 2020;93:208–10.CrossRefPubMed Liang X, Si L, Li Y, Zhang J, Deng J, Bai J, et al. Talaromyces marneffei infection relapse presenting as osteolytic destruction followed by suspected nontuberculous mycobacterium infection during 6 years of follow-up: a case update. Int J Infect Dis. 2020;93:208–10.CrossRefPubMed
5.
go back to reference Wu S, Guo T, Zhang H, He Z, Zhang J, Zeng W. Brain nocardiosis and pulmonary talaromycosis infection in a patient with Anti-IFN-γ autoantibodies: a Case Report. Infect Drug Resist. 2023;16:5421–5.CrossRefPubMedPubMedCentral Wu S, Guo T, Zhang H, He Z, Zhang J, Zeng W. Brain nocardiosis and pulmonary talaromycosis infection in a patient with Anti-IFN-γ autoantibodies: a Case Report. Infect Drug Resist. 2023;16:5421–5.CrossRefPubMedPubMedCentral
6.
go back to reference Ouyang Y, Cai S, Liang H, Cao C. Administration of Voriconazole in disseminated Talaromyces (Penicillium) Marneffei infection: a retrospective study. Mycopathologia. 2017;182(5–6):569–75.CrossRefPubMed Ouyang Y, Cai S, Liang H, Cao C. Administration of Voriconazole in disseminated Talaromyces (Penicillium) Marneffei infection: a retrospective study. Mycopathologia. 2017;182(5–6):569–75.CrossRefPubMed
7.
go back to reference Griffith DE, Aksamit T, Brown-Elliott BA, Catanzaro A, Daley C, Gordin F, et al. An official ATS/IDSA statement: diagnosis, treatment, and prevention of nontuberculous mycobacterial diseases. Am J Respir Crit Care Med. 2007;175(4):367–416.CrossRefPubMed Griffith DE, Aksamit T, Brown-Elliott BA, Catanzaro A, Daley C, Gordin F, et al. An official ATS/IDSA statement: diagnosis, treatment, and prevention of nontuberculous mycobacterial diseases. Am J Respir Crit Care Med. 2007;175(4):367–416.CrossRefPubMed
8.
go back to reference Ma X, Nakayamada S, Kubo S, Sakata K, Yamagata K, Miyazaki Y, et al. Expansion of T follicular helper-T helper 1 like cells through epigenetic regulation by signal transducer and activator of transcription factors. Ann Rheum Dis. 2018;77(9):1354–61.CrossRefPubMed Ma X, Nakayamada S, Kubo S, Sakata K, Yamagata K, Miyazaki Y, et al. Expansion of T follicular helper-T helper 1 like cells through epigenetic regulation by signal transducer and activator of transcription factors. Ann Rheum Dis. 2018;77(9):1354–61.CrossRefPubMed
9.
go back to reference Krisnawati DI, Liu YC, Lee YJ, Wang YT, Chen CL, Tseng PC, et al. Blockade effects of Anti-Interferon- (IFN-) γ autoantibodies on IFN-γ-Regulated antimicrobial immunity. J Immunol Res. 2019;2019:1629258.CrossRefPubMedPubMedCentral Krisnawati DI, Liu YC, Lee YJ, Wang YT, Chen CL, Tseng PC, et al. Blockade effects of Anti-Interferon- (IFN-) γ autoantibodies on IFN-γ-Regulated antimicrobial immunity. J Immunol Res. 2019;2019:1629258.CrossRefPubMedPubMedCentral
10.
go back to reference Kaufman BP, Aman T, Alexis AF. Postinflammatory Hyperpigmentation: Epidemiology, Clinical Presentation, Pathogenesis and Treatment. Am J Clin Dermatol. 2018;19(4):489–503.CrossRefPubMed Kaufman BP, Aman T, Alexis AF. Postinflammatory Hyperpigmentation: Epidemiology, Clinical Presentation, Pathogenesis and Treatment. Am J Clin Dermatol. 2018;19(4):489–503.CrossRefPubMed
11.
go back to reference Hase I, Morimoto K, Sakagami T, Kazumi Y, Ishii Y, van Ingen J. Disseminated Mycobacterium gordonae and Mycobacterium mantenii infection with elevated anti-IFN-γ neutralizing autoantibodies. J Infect Chemother. 2015;21(6):468–72.CrossRefPubMed Hase I, Morimoto K, Sakagami T, Kazumi Y, Ishii Y, van Ingen J. Disseminated Mycobacterium gordonae and Mycobacterium mantenii infection with elevated anti-IFN-γ neutralizing autoantibodies. J Infect Chemother. 2015;21(6):468–72.CrossRefPubMed
12.
go back to reference Aoki A, Sakagami T, Yoshizawa K, Shima K, Toyama M, Tanabe Y, et al. Clinical significance of Interferon-γ neutralizing autoantibodies against disseminated Nontuberculous Mycobacterial Disease. Clin Infect Dis. 2018;66(8):1239–45.CrossRefPubMed Aoki A, Sakagami T, Yoshizawa K, Shima K, Toyama M, Tanabe Y, et al. Clinical significance of Interferon-γ neutralizing autoantibodies against disseminated Nontuberculous Mycobacterial Disease. Clin Infect Dis. 2018;66(8):1239–45.CrossRefPubMed
13.
go back to reference Ning Y, Yu Q, Liang H, Wu S, Liang S, Liang X, et al. Multiple intracellular pathogen infections with ocular pathologies associated with adult-onset immunodeficiency due to anti-interferon-γ autoantibodies: a case report. BMC Infect Dis. 2024;24(1):78.CrossRefPubMedPubMedCentral Ning Y, Yu Q, Liang H, Wu S, Liang S, Liang X, et al. Multiple intracellular pathogen infections with ocular pathologies associated with adult-onset immunodeficiency due to anti-interferon-γ autoantibodies: a case report. BMC Infect Dis. 2024;24(1):78.CrossRefPubMedPubMedCentral
14.
go back to reference Ariyasingha NM, Chowdhury M, Samoilenko A, Salnikov OG, Chukanov NV, Kovtunova LM, et al. Toward Lung Ventilation Imaging using Hyperpolarized Diethyl Ether gas contrast Agent. Chemistry. 2024;30(25):e202304071.CrossRefPubMed Ariyasingha NM, Chowdhury M, Samoilenko A, Salnikov OG, Chukanov NV, Kovtunova LM, et al. Toward Lung Ventilation Imaging using Hyperpolarized Diethyl Ether gas contrast Agent. Chemistry. 2024;30(25):e202304071.CrossRefPubMed
15.
go back to reference Koizumi Y, Sakagami T, Nishiyama N, Hirai J, Hayashi Y, Asai N, et al. Rituximab restores IFN-γ-STAT1 function and ameliorates disseminated Mycobacterium avium infection in a patient with Anti-Interferon-γ Autoantibody. J Clin Immunol. 2017;37(7):644–9.CrossRefPubMed Koizumi Y, Sakagami T, Nishiyama N, Hirai J, Hayashi Y, Asai N, et al. Rituximab restores IFN-γ-STAT1 function and ameliorates disseminated Mycobacterium avium infection in a patient with Anti-Interferon-γ Autoantibody. J Clin Immunol. 2017;37(7):644–9.CrossRefPubMed
16.
go back to reference Zeng W, Tang M, Yang M, Fang G, Tang S, Zhang J. Intravenous cyclophosphamide therapy for Anti-IFN-γ Autoantibody-Associated Talaromyces marneffei infection. Open Forum Infect Dis. 2022;9(12):ofac612.CrossRefPubMedPubMedCentral Zeng W, Tang M, Yang M, Fang G, Tang S, Zhang J. Intravenous cyclophosphamide therapy for Anti-IFN-γ Autoantibody-Associated Talaromyces marneffei infection. Open Forum Infect Dis. 2022;9(12):ofac612.CrossRefPubMedPubMedCentral
Metadata
Title
Multiple pathogen infections accompanied with ocular pathologies in a patient with high-titer Anti-IFN-γ autoantibodies: a case report
Authors
Siyao Wu
Siqiao Liang
Hanlin Liang
Yan Ning
Xia Li
Zhiyi He
Publication date
01-12-2024
Publisher
BioMed Central
Published in
BMC Infectious Diseases / Issue 1/2024
Electronic ISSN: 1471-2334
DOI
https://doi.org/10.1186/s12879-024-10217-2

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