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A case report of Covid-19 in an autoimmune pulmonary alveolar proteinosis: An association in tune with the times!

Autoimmune pulmonary alveolar proteinosis (PAP) is a rare disease characterized by the alveoli accumulation of surfactants proteins and lipids, which diagnosis is confirmed by the presence of GM-CSF antibodies in serum. PAP can be evoked when its characteristic images on chest computed-tomography (C...

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Autores principales: Coirier, Valentin, Delamaire, Flora, Chauvin, Pierre, Kerjouan, Mallorie, Lederlin, Mathieu, Maamar, Adel, Jouneau, Stéphane
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Elsevier 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9969748/
https://www.ncbi.nlm.nih.gov/pubmed/36874265
http://dx.doi.org/10.1016/j.rmcr.2023.101825
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author Coirier, Valentin
Delamaire, Flora
Chauvin, Pierre
Kerjouan, Mallorie
Lederlin, Mathieu
Maamar, Adel
Jouneau, Stéphane
author_facet Coirier, Valentin
Delamaire, Flora
Chauvin, Pierre
Kerjouan, Mallorie
Lederlin, Mathieu
Maamar, Adel
Jouneau, Stéphane
author_sort Coirier, Valentin
collection PubMed
description Autoimmune pulmonary alveolar proteinosis (PAP) is a rare disease characterized by the alveoli accumulation of surfactants proteins and lipids, which diagnosis is confirmed by the presence of GM-CSF antibodies in serum. PAP can be evoked when its characteristic images on chest computed-tomography (CT) are present: bilateral and multifocal ground-glass opacities and crazy-paving appearance. Patients with PAP are at an increased risk of opportunistic infections caused by Nocardia, mycobacteria and fungal pathogens due to impaired processing of pulmonary surfactant. We here report a typical case of newly diagnosed autoimmune PAP, with initial indication to realize a whole-lung lavage. Despite this treatment the patient presented a marked clinical worsening, with increasing need for oxygen and finally the need for mechanical ventilation. The chest CT was controlled and found to be typical of PAP, while the search for opportunistic infections remained negative. Finally, SARS-CoV-2 PCR was performed on bronchoalveolar lavage fluid, and was positive, whereas it had previously been negative twice. Our case report highlights the difficulty of distinguishing SARS-CoV-2 infection in the context of PAP, as the chest CT features are similar. We believe that a SARS-CoV-2 RT-PCR should be systematically realized in case of respiratory deterioration in PAP patients.
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spelling pubmed-99697482023-02-27 A case report of Covid-19 in an autoimmune pulmonary alveolar proteinosis: An association in tune with the times! Coirier, Valentin Delamaire, Flora Chauvin, Pierre Kerjouan, Mallorie Lederlin, Mathieu Maamar, Adel Jouneau, Stéphane Respir Med Case Rep Case Report Autoimmune pulmonary alveolar proteinosis (PAP) is a rare disease characterized by the alveoli accumulation of surfactants proteins and lipids, which diagnosis is confirmed by the presence of GM-CSF antibodies in serum. PAP can be evoked when its characteristic images on chest computed-tomography (CT) are present: bilateral and multifocal ground-glass opacities and crazy-paving appearance. Patients with PAP are at an increased risk of opportunistic infections caused by Nocardia, mycobacteria and fungal pathogens due to impaired processing of pulmonary surfactant. We here report a typical case of newly diagnosed autoimmune PAP, with initial indication to realize a whole-lung lavage. Despite this treatment the patient presented a marked clinical worsening, with increasing need for oxygen and finally the need for mechanical ventilation. The chest CT was controlled and found to be typical of PAP, while the search for opportunistic infections remained negative. Finally, SARS-CoV-2 PCR was performed on bronchoalveolar lavage fluid, and was positive, whereas it had previously been negative twice. Our case report highlights the difficulty of distinguishing SARS-CoV-2 infection in the context of PAP, as the chest CT features are similar. We believe that a SARS-CoV-2 RT-PCR should be systematically realized in case of respiratory deterioration in PAP patients. Elsevier 2023-02-27 /pmc/articles/PMC9969748/ /pubmed/36874265 http://dx.doi.org/10.1016/j.rmcr.2023.101825 Text en © 2023 The Authors https://creativecommons.org/licenses/by/4.0/This is an open access article under the CC BY license (http://creativecommons.org/licenses/by/4.0/).
spellingShingle Case Report
Coirier, Valentin
Delamaire, Flora
Chauvin, Pierre
Kerjouan, Mallorie
Lederlin, Mathieu
Maamar, Adel
Jouneau, Stéphane
A case report of Covid-19 in an autoimmune pulmonary alveolar proteinosis: An association in tune with the times!
title A case report of Covid-19 in an autoimmune pulmonary alveolar proteinosis: An association in tune with the times!
title_full A case report of Covid-19 in an autoimmune pulmonary alveolar proteinosis: An association in tune with the times!
title_fullStr A case report of Covid-19 in an autoimmune pulmonary alveolar proteinosis: An association in tune with the times!
title_full_unstemmed A case report of Covid-19 in an autoimmune pulmonary alveolar proteinosis: An association in tune with the times!
title_short A case report of Covid-19 in an autoimmune pulmonary alveolar proteinosis: An association in tune with the times!
title_sort case report of covid-19 in an autoimmune pulmonary alveolar proteinosis: an association in tune with the times!
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9969748/
https://www.ncbi.nlm.nih.gov/pubmed/36874265
http://dx.doi.org/10.1016/j.rmcr.2023.101825
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