Unusual Presentation of Severe Endobronchial Obstruction Caused by Cryptococcus gattii in a Child

Huynh, Julie; Saddi, Vishal; Cooper, Peter; Cheng, Alan; Meyer, Weiland; Chen, Sharon; Isaacs, David

doi:10.1093/jpids/piy100

Cited by 11 publications

(9 citation statements)

References 27 publications

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“…These infections may be secondary to PAP, the use of steroids, and/or impaired GM-CSF signaling directly compromising alveolar macrophage immunity to these pathogens ( Punatar et al, 2012 ). Since 2013, high titers of IgG (mostly IgG1) auto-Abs neutralizing 10 ng/ml GM-CSF (plasma diluted 1/10) have been found in patients with adult-onset isolated idiopathic disseminated disease mostly due to Cryptococcus spp., almost exclusively Cryptococcus gattii ( Applen Clancey et al, 2019 ; Browne et al, 2012a ; Crum-Cianflone et al, 2017 ; Demir et al, 2018 ; Huynh et al, 2020 ; Kuo et al, 2017 ; Panackal et al, 2017 ; Perrineau et al, 2020 ; Rosen et al, 2013 ; Saijo et al, 2014 ; Stevenson et al, 2019 ; Viola et al, 2021 ), Nocardia spp. ( Rosen et al, 2015 ), or more rarely, Aspergillus spp.…”

Section: Neutralizing Auto-abs Against Gm-csfmentioning

confidence: 99%

Human autoantibodies underlying infectious diseases

Puel

Bastard

Bustamante

et al. 2022

Journal of Experimental Medicine

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The vast interindividual clinical variability observed in any microbial infection—ranging from silent infection to lethal disease—is increasingly being explained by human genetic and immunological determinants. Autoantibodies neutralizing specific cytokines underlie the same infectious diseases as inborn errors of the corresponding cytokine or response pathway. Autoantibodies against type I IFNs underlie COVID-19 pneumonia and adverse reactions to the live attenuated yellow fever virus vaccine. Autoantibodies against type II IFN underlie severe disease caused by environmental or tuberculous mycobacteria, and other intra-macrophagic microbes. Autoantibodies against IL-17A/F and IL-6 are less common and underlie mucocutaneous candidiasis and staphylococcal diseases, respectively. Inborn errors of and autoantibodies against GM-CSF underlie pulmonary alveolar proteinosis; associated infections are less well characterized. In individual patients, autoantibodies against cytokines preexist infection with the pathogen concerned and underlie the infectious disease. Human antibody-driven autoimmunity can interfere with cytokines that are essential for protective immunity to specific infectious agents but that are otherwise redundant, thereby underlying specific infectious diseases.

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Section: Neutralizing Auto-abs Against Gm-csfmentioning

confidence: 99%

Human autoantibodies underlying infectious diseases

Puel

Bastard

Bustamante

et al. 2022

Journal of Experimental Medicine

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“…Demir et al [29 ▪ ] presented a 42-year-old male with C. gattii meningitis and anti-GM-CSF autoantibodies and in whom PAP developed 3 years later. Huynh et al [30] reported another unusual presentation of endobronchial lesions due to C. gattii in the presence of anti-GM-CSF autoantibodies although their inhibitory activity was not formally ascertained. Clancey et al , recently described an interesting case of travel-acquired anti-GM-CSF associated C. deuterogattii meningitis in an otherwise immunocompetent 69-year-old man from North Carolina which occurred 3 month after he experienced a period of cough and shortness of breath.…”

Section: Antibodies To Granulocyt Macrophage Colony-stimulating Factomentioning

confidence: 99%

Anticytokine autoantibodies leading to infection: early recognition, diagnosis and treatment options

Barcenas-Morales

Cortes-Acevedo

Döffinger

2019

Current Opinion in Infectious Diseases

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Purpose of review The current review gives a concise and updated overview of the relative new field of anticytokine autoantibodies (ACAA) and associated infections with a focus on recent findings regarding clinical manifestions, diagnostic and treatments. Recent findings Several recent case reports of unusual presentations of patients with neutralizing autoantibodies to IFN-γ and granulocyt macrophage colony-stimulating factor and expand the spectrum of clinical manifestations and suggest that anticytokine-mediated acquired immunodeficiency causing susceptibility to infection may be underdiagnosed. There is an expanding geographical distribution of antigranulocyt macrophage colony-stimulating factor associated Cryptococcus gattii infection. The spectrum of identified infections in patients with neutralizing antibodies to IFN-γ has a strong endemic component. Rituximab or cyclophophamide in addition to antimycobacterials could be a treatment options in refractory cases. NF-κB2 deficiency may be associated with a complex pattern of high titre neutralizing ACAA similar to autoimmune polyglandular syndrome type I and Thymoma. New technique for the detection of anticytokine antibodies are presented. Quantiferon testing, which is widely available for TB-diagnostic, may be repurposed to detect anti-IFN-γ autoantibodies. We propose that this test could be as well used to show if they are neutralizing. Summary ACAA are an emerging cause of acquired immunodeficiency which is likely underdiagnosed. Recent case reports document expanding spectra of clinical manifestations. NF-κB2 deficiency may be associated with a complex anti cytokine autoantibody pattern.

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“…Illustrating the principal that severe fungal disease is not fundamentally congruent with an “immunocompetent” state, further immunological investigations were pursued, revealing aab:GM‐CSF in HIV‐uninfected individuals in such patients 215,216 . That aab:GM‐CSF predispose to CNS and other forms of disseminated cryptococcosis, and less frequently to exclusive pulmonary disease, in individuals without a known congenital or acquired immunodeficiency has been confirmed by numerous groups globally 217–225 . While aab:GM‐CSF have been overwhelmingly associated with C. gattii , they may also underlie disease due to C. neoformans 226–232 .…”

Section: Cryptococcosismentioning

confidence: 99%

“…215,216 That aab:GM-CSF predispose to CNS and other forms of disseminated cryptococcosis, and less frequently to exclusive pulmonary disease, in individuals without a known congenital or acquired immunodeficiency has been confirmed by numerous groups globally. [217][218][219][220][221][222][223][224][225] While aab:GM-CSF have been overwhelmingly associated with C. gattii, they may also underlie disease due to C. neoformans. [226][227][228][229][230][231][232] Notably, aab:GM-CSF can predispose to infection in the absence or predating the development of PAP 215,228 ; thus, the absence of PAP does not exclude the possibility of aab:GM-CSF as a cause of fungal susceptibility.…”

Section: Cryp Tococcosismentioning

confidence: 99%

From Mendel to mycoses: Immuno‐genomic warfare at the human–fungus interface

Vinh

2023

Immunological Reviews

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SummaryFungi are opportunists: They particularly require a defect of immunity to cause severe or disseminated disease. While often secondary to an apparent iatrogenic cause, fungal diseases do occur in the absence of one, albeit infrequently. These rare cases may be due to an underlying genetic immunodeficiency that can present variably in age of onset, severity, or other infections, and in the absence of a family history of disease. They may also be due to anti‐cytokine autoantibodies. This review provides a background on how human genetics or autoantibodies underlie cases of susceptibility to severe or disseminated fungal disease. Subsequently, the lessons learned from these inborn errors of immunity marked by fungal disease (IEI‐FD) provide a framework to begin to mechanistically decipher fungal syndromes, potentially paving the way for precision therapy of the mycoses.

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Unusual Presentation of Severe Endobronchial Obstruction Caused by Cryptococcus gattii in a Child

Cited by 11 publications

References 27 publications

Human autoantibodies underlying infectious diseases

Human autoantibodies underlying infectious diseases

Anticytokine autoantibodies leading to infection: early recognition, diagnosis and treatment options

From Mendel to mycoses: Immuno‐genomic warfare at the human–fungus interface

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