title: Clinical manifestations, course, and outcome of patients with neutralizing anti-interferon-γ autoantibodies and disseminated nontuberculous mycobacterial infections.
abstract: Neutralizing anti-interferon-γ autoantibody (nAIGA)-associated immunodeficiency is an emerging medical issue worldwide. In the present study, we describe and discuss the clinical features and outcomes of patients with nAIGAs and disseminated infections by nontuberculous mycobacteria (dNTM).We thoroughly reviewed the medical records of all patients. Microorganisms and nAIGAs were identified using previously described methods with modifications. All data were calculated and analyzed using SPSS software.Among 46 adult patients with dNTM infections, we identified 45 cases (97.8%) with nAIGAs. The average patient age was 58.6 years, and there was no sex predominance. Cervical lymphadenitis (81.8%) was the most common clinical manifestation. Endocrine disorder was the leading comorbidity (7 cases). Malignancies were found in 4 patients, and all of the malignancies originated from the T-cell/macrophage lineage. More than half of the identifiable isolates were slow-growing NTMs. Twenty-eight (62.2%) and 18 (40.0%) patients had a history of zoster and salmonellosis, respectively. A high proportion of patients with recurrent episodes of NTM infection or a history of zoster and dNTM infection had initial nAIGA titers ≥10 dilution (P < 0.05). Twenty-seven patients (60.0%) required long-term antimycobacterial therapy and had at least 1 episode of recurrent NTM disease. No mortality was related to dNTM infection.In Taiwan, nAIGAs are a recently recognized mechanism of dNTM infection. Long term of antibiotic treatment and adherence to medical advice are necessary to improve the clinical outcome of patients with nAIGAs.
authors.affiliation: Laboratory of Human Immunology and Infectious Diseases, Graduate Institute of Clinical Medical Sciences, Chang Gung University, Taoyuan, Taiwan Division of Infectious Diseases, Department of Internal Medicine, China Medical University Hospital, Taichung, Taiwan School of Medicine, College of Medicine, China Medical University, Taichung, Taiwan Graduate Institute of Biomedical Science, Chang Gung University, Taoyuan, Taiwan Division of Infectious Diseases, Department of Internal Medicine, Chang Gung Memorial Hospital, Kaohsiung, Taiwan Division of Infectious Diseases, Department of Internal Medicine, Chang Gung Memorial Hospital, Taoyuan, Taiwan Division of Infectious Diseases, Department of Internal Medicine, Taipei Veterans General Hospital, Taipei, Taiwan Division of Infectious Diseases, Department of Internal Medicine, National Taiwan University Hospital, Taipei, Taiwan Division of Infectious Diseases, Department of Internal Medicine, Changhua Christian Hospital, Changhua, Taiwan Department of Chest Medicine, Taipei Veterans General Hospital, Taipei, Taiwan Department of Internal Medicine, Buddhist Tzu Chi General Hospital, Hualien, Taiwan Department of Infectious Diseases, Buddhist Tzu Chi General Hospital, Hualien, Taiwan Department of Orthopedics, China Medical University Hospital, Taichung, Taiwan Division of Infectious Diseases, Department of Internal Medicine, Chang Gung Memorial Hospital, Chiayi, Taiwan Division of Pulmonary and Critical Care Medicine, Department of Internal Medicine, China Medical University Hospital, Taichung, Taiwan Division of Infectious Disease, Department of Internal Medicine, Buddhist Tzu Chi General Hospital, Taichung, Taiwan Division of Infectious Diseases, Koo Foundation Sun Yat-Sen Cancer Center, Taipei, Taiwan Department of Trauma and Emergency Surgery, Chang Gung Memorial Hospital, Linkou, Taoyuan, Taiwan Division of Infectious Diseases, Department of Pediatrics, Chang Gung Memorial Hospital, Taoyuan, Taiwan Graduate Institute of Clinical Medical Science, China Medical University, Taichung, Taiwan Chang Gung Immunology Consortium, Chang Gung Memorial Hospital and Chang Gung University, Taoyuan, Taiwan.
authors.forename: Chih-Yu
authors.initials: CY
authors.lastname: Chi
authors.forename: Chia-Hao
authors.initials: CH
authors.lastname: Lin
authors.forename: Mao-Wang
authors.initials: MW
authors.lastname: Ho
authors.forename: Jing-Ya
authors.initials: JY
authors.lastname: Ding
authors.forename: Wen-Chi
authors.initials: WC
authors.lastname: Huang
authors.forename: Han-Po
authors.initials: HP
authors.lastname: Shih
authors.forename: Chun-Fu
authors.initials: CF
authors.lastname: Yeh
authors.forename: Chang-Phone
authors.initials: CP
authors.lastname: Fung
authors.forename: Hsin-Yun
authors.initials: HY
authors.lastname: Sun
authors.forename: Ching-Tai
authors.initials: CT
authors.lastname: Huang
authors.forename: Ting-Shu
authors.initials: TS
authors.lastname: Wu
authors.forename: Chih-Yen
authors.initials: CY
authors.lastname: Chang
authors.forename: Yuag-Meng
authors.initials: YM
authors.lastname: Liu
authors.forename: Jia-Yih
authors.initials: JY
authors.lastname: Feng
authors.forename: Wei-Kai
authors.initials: WK
authors.lastname: Wu
authors.forename: Lih-Shinn
authors.initials: LS
authors.lastname: Wang
authors.forename: Chung-Hao
authors.initials: CH
authors.lastname: Tsai
authors.forename: Cheng-Mao
authors.initials: CM
authors.lastname: Ho
authors.forename: Huang-Shen
authors.initials: HS
authors.lastname: Lin
authors.forename: Hung-Jen
authors.initials: HJ
authors.lastname: Chen
authors.forename: Po-Chang
authors.initials: PC
authors.lastname: Lin
authors.forename: Wei-Chin
authors.initials: WC
authors.lastname: Liao
authors.forename: Wei-Ting
authors.initials: WT
authors.lastname: Chen
authors.forename: Chia-Chi
authors.initials: CC
authors.lastname: Lo
authors.forename: Shang-Yu
authors.initials: SY
authors.lastname: Wang
authors.forename: Chen-Yen
authors.initials: CY
authors.lastname: Kuo
authors.forename: Chen-Hsiang
authors.initials: CH
authors.lastname: Lee
authors.forename: Cheng-Lung
authors.initials: CL
authors.lastname: Ku
mesh_terms: D000328:Adult; D000368:Aged; D000369:Aged, 80 and over; D000900:Anti-Bacterial Agents / Q000627:therapeutic use*; D000888:Antibodies, Anti-Idiotypic / Q000276:immunology*; D001323:Autoantibodies / Q000276:immunology*; D005260:Female; D005500:Follow-Up Studies; D006801:Humans; D015994:Incidence; D007371:Interferon-gamma / Q000276:immunology*; D008297:Male; D008875:Middle Aged; D009165:Mycobacterium Infections, Nontuberculous / Q000188:drug therapy / Q000453:epidemiology* / Q000276:immunology; D009170:Nontuberculous Mycobacteria / Q000276:immunology*; D011379:Prognosis; D012189:Retrospective Studies; D013624:Taiwan / Q000453:epidemiology; D013997:Time Factors
chemical_list: D000900:Anti-Bacterial Agents; D000888:Antibodies, Anti-Idiotypic; D001323:Autoantibodies; D007371:Interferon-gamma