Factors Associated With Acquired Anti-Interferon-gamma (IFN-γ) Autoantibody in Patients With Nontuberculous Mycobacterial Infection

Phoompoung, Pakpoom; Ankasekwinai, Nasikarn; Suputtamongkol, Yupin

doi:10.1093/ofid/ofw172.438

Cited by 3 publications

(5 citation statements)

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“…Our finding that peripheral lymph nodes were the most commonly infected sites in patients with neutralizing anti-IFNg Abs is consistent with the observations in other cohort studies, regardless of ethnicities and causative microorganisms [3,11,12,14]. The laboratory feature of marked leukocytosis is also consistent with the observations in two retrospective caseecontrol studies [3,15] as well as in other case reports of neutralizing anti-IFNg Abs [4,9,16,17]. However, this feature is in contrast to the known complications of bone marrow suppression and pancytopenia caused by dNTM associated with other immunodeficiencies [18e20], which are attributed to underlying mechanisms such as myelofibrosis, haemophagocytosis and depletion of haematopoietic progenitors induced by chronic infection [21,22].…”

Section: Discussionsupporting

confidence: 92%

“…Our study had some limitations, including the small sample size and limited generalizability of the findings to patients with other opportunistic infections because all of our case patients had NTM infection. However, while the majority of previous studies of neutralizing anti-IFNg Abs were case series or included a heterogeneous population of localized and disseminated NTM infections caused by different underlying mechanisms [1,3,15], only otherwise healthy individuals with disseminated NTM infection were included in our caseecontrol analysis. In addition, because our study was conducted prospectively, we could obtain the earliest and the most accurate clinical information at disease onset and minimize the interference caused by subsequent treatment.…”

Section: Discussionmentioning

confidence: 99%

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Incorrect diagnoses in patients with neutralizing anti-interferon-gamma-autoantibodies

Wang

Sheng

et al. 2020

Clinical Microbiology and Infection

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Disseminated non-tuberculous mycobacterial infections Incorrect diagnosesNeutralizing anti-interferon-gammaautoantibodies TB metastatic carcinoma a b s t r a c t Objectives: Early diagnosis of adult-onset immunodeficiency associated with neutralizing antiinterferon-gamma autoantibodies (anti-IFNg Abs) remains difficult given the lack of a distinctive phenotype and a routine test. This study aimed to investigate the determinants of incorrect tentative diagnoses and useful clues for early disease recognition. Methods: This study enrolled adult patients who had unexplained opportunistic infections diagnosed at six hospitals and identified those having neutralizing anti-IFNg Abs (cases). Demographics, medical history, initial presentations and laboratory data, causative pathogens, tentative diagnoses, and treatment were analysed and compared among individuals having neutralizing anti-IFNg Abs (cases) and those without (controls). Results: Among the 154 patients enrolled, neutralizing anti-IFN-g Abs were detected in 50 (71%) of 70 patients with disseminated non-tuberculous mycobacterial infection (dNTM) but not in 84 patients without dNTM. The median time from disease onset to the recognition of dNTM associated with neutralizing anti-IFNg Abs was 1.6 years (range, 0.25e19 years). Incorrect tentative diagnoses resulted in the administration of anti-tuberculosis regimens (60%, 30/50), immunosuppressants (48%, 24/50), and systemic chemotherapy (2%, 10/50) to the 50 cases. Multivariate analysis revealed that case patients were more likely than controls to present with multiple bone lesions (adjusted odds ratio (OR), 27.16; 95% confidence interval (CI), 1.21e609.59) and leukocytosis (adjusted OR, 1.48; 95% CI, 1.12e1.95); however, the controls had a higher rate of mycobacterial bloodstream infection (adjusted OR, 0.05; 95% CI 0.00 e0.66). Conclusions: The high rate of incorrect tentative diagnoses led to frequent inappropriate management in patients with neutralizing anti-IFNg Abs, and highlighted the need for increased awareness among clinicians. U.-I.

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Section: Discussionsupporting

confidence: 92%

Section: Discussionmentioning

confidence: 99%

Incorrect diagnoses in patients with neutralizing anti-interferon-gamma-autoantibodies

Wang

Sheng

et al. 2020

Clinical Microbiology and Infection

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“…Thus, when dermatologists encounter patients affected with the syndrome who present with ND, one should specifically search for opportunistic infections that may be occult at that state. Cutaneous involvements have been reported in 49-57% of patients in other case series (3,14,15). Type of reactive dermatoses keep with our findings, being mainly ND.…”

Section: Discussionsupporting

confidence: 90%

“…Moreover, it is rarely reported in HIV infected individuals (26)(27)(28). Until recently, ND has been reported to be found in NTM patients affected by this syndrome (1,3,14,15). The exact pathogenesis of ND remains to be elucidated.…”

Section: Discussionmentioning

confidence: 99%

Skin Manifestations in Patients with Adult-onset Immunodeficiency due to Anti-interferon-gamma Autoantibody: A Relationship with Systemic Infections

Jutivorakool¹,

Sittiwattanawong²,

Kantikosum³

et al. 2018

Acta Derm Venerol

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Adult-onset immunodeficiency due to anti-interferon-γ autoantibody is an emerging acquired immunodeficiency with frequent skin manifestations. A retrospective chart review was conducted and identified 41 patients with the syndrome. Skin involvement was detected in 33 (80%) patients, 15 (45%) with infective skin diseases and 27 (82%) with reactive skin disorders. Reactive lesions were mostly neutrophilic dermatoses, e.g. Sweet syndrome. Of note, the presence of neutrophilic dermatoses was highly associated with infections of other sites. An adjusted odds ratio for the existence of infections in patients with neutrophilic dermatoses was 14.79 (95% CI: 5.13, 42.70; p < 0.001). Moreover, neutrophilic dermatoses were significantly correlated with opportunistic infections observed in those with defects in cell-mediated immunity including non-tuberculous mycobacterium and disseminated fungal infection. The odds ratio for opportunistic infections in the presence of neutrophilic dermatoses was 12.35 (95% CI: 5.00, 30.55; p <0.001). Thus, the presence of neutrophilic dermatoses in patients with the syndrome can signal opportunistic infections that warrant physician attention.

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“…1,2,6 Bone marrow or blood, lung, bladder, liver, and biliary tree are likewise the sites of infection. 6,24–26 The clinical manifestations, locations of the infected sites, and causative agents are likely to differ across ethnicities. Rapidly growing mycobacteria (RGM), such as M. abscessus , were the most common NTM species isolated from Thai, Chinese and Filipino patients, whereas M. avium complex (MAC) was predominant among Japanese and non-Asian patients.…”

Section: Anti-ifn-γ Aabs and Pathologymentioning

confidence: 99%

Minireview: Insights into anti-interferon-γ autoantibodies

Chawansuntati

Rattanathammethee

Wipasa

2021

Exp Biol Med (Maywood)

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The association between the presence of anti-interferon-γ autoantibodies and the onset of immunodeficiency with intracellular infections has been clearly established. No standard regimen to control the production of these pathogenic autoantibodies, apart from antimicrobial therapy to eliminate infections, contributes to the medical burden of this syndrome, which sometimes has a fatal outcome. In this review, we summarize the findings on anti-interferon-γ autoantibodies to facilitate further research and to provide guidance for treatment strategies.

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Factors Associated With Acquired Anti-Interferon-gamma (IFN-γ) Autoantibody in Patients With Nontuberculous Mycobacterial Infection

Cited by 3 publications

References 0 publications

Incorrect diagnoses in patients with neutralizing anti-interferon-gamma-autoantibodies

Incorrect diagnoses in patients with neutralizing anti-interferon-gamma-autoantibodies

Skin Manifestations in Patients with Adult-onset Immunodeficiency due to Anti-interferon-gamma Autoantibody: A Relationship with Systemic Infections

Minireview: Insights into anti-interferon-γ autoantibodies

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