Pulmonary alveolar proteinosis following cryptococcal meningitis: a possible cause?

Demir, Sarah; Chebib, Nader; Thivolet‐Béjui, F.; Cottin, Vincent

doi:10.1136/bcr-2017-222940

Cited by 17 publications

(13 citation statements)

References 8 publications

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“…Patient plasmas were shown to inhibit GM-CSF-induced STAT5 phosphorylation [28]. 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.…”

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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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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“…Based on our literature search, thus far, there is only one case report from France that reported a case of confirmed autoimmune PAP preceded by Cryptococcus gattii meningitis with positive anti-GM-CSF antibody [19]. Two similar cases were reported from America with Cryptococcus neoformans.…”

Section: Discussionmentioning

confidence: 99%

“…Anti GM-CSF antibodies were positive in high titre. He was not offered any specific treatment and three years after the diagnosis of PAP patient remained asymptomatic with near complete resolution of radiological lesion and decrease in anti-GM-CSF antibody titre [19].…”

Section: Casementioning

confidence: 99%

Crazy-Paving Pattern: A Rare Case of Autoimmune Pulmonary Alveolar Proteinosis (PAP) with Positive Anti-GM-CSF Antibody Following Cryptococcal Infection in an otherwise Healthy Individual and Review of Literature

2021

Eur J Respir Med

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Pulmonary alveolar proteinosis (PAP) is a rare disease characterised by accumulation of lipoproteinaceous material composed principally of surfactant phospholipid and apoproteins within the alveoli, resulting in impairment of gas exchange [1,2]. It was first described by Rosen et al. in 1958 [1]. There is a wide range of clinical manifestation of PAP ranging from respiratory failure to spontaneous resolution.Autoimmune PAP is characterised by positive anti-GM-CSF antibody and presence of this antibody increase the susceptibility to opportunistic infections. We report a rare case of a middle-aged reformed smoker who was diagnosed with autoimmune PAP six years following his initial diagnosis of cryptococcal meningitis and pulmonary cryptoccocoma.

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“…Nowadays, reports of OIs in patients with confirmed anti-GM-CSF autoantibodies with or without PAP have been increasing. 8,15,18,[23][24][25][26][27][28][29][30] Since the first documented case of anti-GM-CSF autoantibodies associated with disseminated cryptococcosis, 15 a total of 27 cases have been reported in the literature (Table 1). The median age at the time of presentation was 41 years (range 20-73 years).…”

Section: A S E Discussion and Re Vie W Of The Liter Aturementioning

confidence: 99%

Disseminated cryptococcosis and anti‐granulocyte‐macrophage colony‐stimulating factor autoantibodies: An underappreciated association

Viola

Malek

Rosen

et al. 2021

Mycoses

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The development of disseminated cryptococcosis has historically occurred in patients living with advanced human immunodeficiency virus or other immunosuppressive conditions affecting T‐cell function. Recently, patients with anti‐cytokine neutralising autoantibodies have been recognised to be at risk for disseminated infections by opportunistic intracellular pathogens, including Cryptococcus species. Herein, we present a previously healthy 26‐year‐old man who was evaluated with disseminated cryptococcosis involving the bone, lung, mediastinum and brain. The patient's serum cryptococcal antigen titres were >1:1,100,000, and evaluation for an underlying immunodeficiency revealed high titres for anti‐granulocyte‐macrophage colony‐stimulating factor (GM‐CSF) autoantibodies. We also review the literature of all published cases of disseminated cryptococcosis associated with the presence of anti‐GM‐CSF autoantibodies. Clinicians should have a heightened awareness of anti‐cytokine autoantibodies in patients without a known immunodeficiency and development disseminated infections by opportunistic intracellular pathogens.

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Pulmonary alveolar proteinosis following cryptococcal meningitis: a possible cause?

Cited by 17 publications

References 8 publications

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

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

Crazy-Paving Pattern: A Rare Case of Autoimmune Pulmonary Alveolar Proteinosis (PAP) with Positive Anti-GM-CSF Antibody Following Cryptococcal Infection in an otherwise Healthy Individual and Review of Literature

Disseminated cryptococcosis and anti‐granulocyte‐macrophage colony‐stimulating factor autoantibodies: An underappreciated association

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