Multiple refractory intracellular pathogen infections in a human immunodeficiency virus-negative patient with anti-interferon-γ autoantibodies: a case report
-
Published:2023-07-26
Issue:1
Volume:23
Page:
-
ISSN:1471-2334
-
Container-title:BMC Infectious Diseases
-
language:en
-
Short-container-title:BMC Infect Dis
Author:
Wang Hongxia,Lei Rong,Ji Yang,Xu Wei,Zhang Keke,Guo Xiang
Abstract
Abstract
Background
The clinical presentation of adult-onset immunodeficiency with anti-interferon (IFN)-γ autoantibodies with intracellular pathogens can be highly variable, which can lead to misdiagnosis during the early stage of disease.
Case presentation
We report a complex case of a 54-year-old Chinese male who was human immunodeficiency virus-negative. He had a presence of anti-IFN-γ autoantibodies and suffered from various intracellular pathogenic infections. The patient was admitted to our hospital for the first time in July 2016 with severe pneumonia, and he experienced multiple pneumonia infections between 2017 and 2019. In March 2019, the patient was hospitalized due to pulmonary lesions and multiple-bone destruction. During hospitalization, the patient was confirmed to have disseminated Talaromyces marneffei infection and was successfully treated with antifungal therapy for 1 year. In June 2021, Mycobacterium kansasii infection was detected by positive culture and progressive bone destruction. A high concentration of anti-IFN-γ antibodies was observed in the patient’s serum. In addition, Listeria monocytogenes was isolated by blood culture, and the presence of L. monocytogenes in cerebrospinal fluid was confirmed by next-generation sequencing. Following anti-non-tuberculous mycobacteria (NTM) therapy and anti-bacterial therapy, the patient’s symptoms, pulmonary lesions, and bone destruction gradually improved.
Conclusions
Although the clinical presentation of adult-onset immunodeficiency with anti-IFN-γ autoantibodies can be highly variable, the diagnosis should be considered if patients suffer from unexplained repeated bacterial or opportunistic infections.
Conventional and advanced molecular testing should be used, as needed, for microbiological diagnoses among this special immunodeficient population.
Publisher
Springer Science and Business Media LLC
Subject
Infectious Diseases
Reference26 articles.
1. Döffinger R, Helbert MR, Barcenas-Morales G, Yang K, Dupuis S, Ceron-Gutierrez L, Espitia-Pinzon C, Barnes N, Bothamley G, Casanova JL, Longhurst HJ, Kumararatne DS. Autoantibodies to interferon-gamma in a patient with selective susceptibility to mycobacterial infection and organ-specific autoimmunity. Clin Infect Dis. 2004;38(1):e10–4. 2. Browne SK, Burbelo PD, Chetchotisakd P, Suputtamongkol Y, Kiertiburanakul S, Shaw PA, Kirk JL, Jutivorakool K, Zaman R, Ding L, Hsu AP, Patel SY, Olivier KN, Lulitanond V, Mootsikapun P, Anunnatsiri S, Angkasekwinai N, Sathapatayavongs B, Hsueh PR, Shieh CC, Brown MR, Thongnoppakhun W, Claypool R, Sampaio EP, Thepthai C, Waywa D, Dacombe C, Reizes Y, Zelazny AM, Saleeb P, Rosen LB, Mo A, Iadarola M, Holland SM. Adult-onset immunodeficiency in Thailand and Taiwan. N Engl J Med. 2012;367(8):725–34. 3. Xie YL, Rosen LB, Sereti I, Barber DL, Chen RY, Hsu DC, Qasba SS, Zerbe CS, Holland SM, Browne SK. Severe paradoxical reaction during treatment of disseminated tuberculosis in a patient with neutralizing anti-IFNγ autoantibodies. Clin Infect Dis. 2016;62(6):770–3. 4. Wipasa J, Chaiwarith R, Chawansuntati K, Praparattanapan J, Rattanathammethee K, Supparatpinyo K. Characterization of anti-interferon-γ antibodies in HIV-negative immunodeficient patients infected with unusual intracellular microorganisms. Exp Biol Med (Maywood). 2018;243(7):621–6. 5. Liang X, Si L, Li Y, Zhang J, Deng J, Bai J, Li M, He Z. 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.
Cited by
1 articles.
订阅此论文施引文献
订阅此论文施引文献,注册后可以免费订阅5篇论文的施引文献,订阅后可以查看论文全部施引文献
|
|