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Response to inhaled granulocyte‐macrophage colony‐stimulating factor in patient with mild‐to‐moderate autoimmune pulmonary alveolar proteinosis—24 months of follow‐up

INTRODUCTION: Autoimmune pulmonary alveolar proteinosis (aPAP) is a disease caused by IgG antibodies against granulocyte‐macrophage colony‐stimulating factor (GM‐CSF). Whole lung lavage (WLL) allows to remove the lipo‐proteinaceous material accumulated by the poor clearance of alveolar surfactant. H...

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Autores principales: Oterino‐Moreira, Iván, Linares‐Asensio, María‐Jesús, Sanz‐Márquez, Sira, Pérez‐Encinas, Montserrat
Formato: Online Artículo Texto
Lenguaje:English
Publicado: John Wiley and Sons Inc. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10542988/
https://www.ncbi.nlm.nih.gov/pubmed/37300395
http://dx.doi.org/10.1111/crj.13650
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author Oterino‐Moreira, Iván
Linares‐Asensio, María‐Jesús
Sanz‐Márquez, Sira
Pérez‐Encinas, Montserrat
author_facet Oterino‐Moreira, Iván
Linares‐Asensio, María‐Jesús
Sanz‐Márquez, Sira
Pérez‐Encinas, Montserrat
author_sort Oterino‐Moreira, Iván
collection PubMed
description INTRODUCTION: Autoimmune pulmonary alveolar proteinosis (aPAP) is a disease caused by IgG antibodies against granulocyte‐macrophage colony‐stimulating factor (GM‐CSF). Whole lung lavage (WLL) allows to remove the lipo‐proteinaceous material accumulated by the poor clearance of alveolar surfactant. However, it is a complex technique that is not exempt from complications, and in some cases, the patients are refractory, requiring the performance of several WLLs spaced apart in time. MATERIALS AND METHODS: We present the clinical, functional, and radiological evolution after 24 months of follow‐up of a patient diagnosis of aPAP refractory to WLL, with performed three therapeutic WLLs spaced 16 and 36 months and serious potentially fatal complications in the last one. RESULTS AND DISSCUSION: After 24 months, no adverse effects have appeared and the great clinical, functional and radiological response is maintained. The patient has been successfully treated with inhaled recombinant human GM‐CSF sargramostim.
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spelling pubmed-105429882023-10-03 Response to inhaled granulocyte‐macrophage colony‐stimulating factor in patient with mild‐to‐moderate autoimmune pulmonary alveolar proteinosis—24 months of follow‐up Oterino‐Moreira, Iván Linares‐Asensio, María‐Jesús Sanz‐Márquez, Sira Pérez‐Encinas, Montserrat Clin Respir J Brief Reports INTRODUCTION: Autoimmune pulmonary alveolar proteinosis (aPAP) is a disease caused by IgG antibodies against granulocyte‐macrophage colony‐stimulating factor (GM‐CSF). Whole lung lavage (WLL) allows to remove the lipo‐proteinaceous material accumulated by the poor clearance of alveolar surfactant. However, it is a complex technique that is not exempt from complications, and in some cases, the patients are refractory, requiring the performance of several WLLs spaced apart in time. MATERIALS AND METHODS: We present the clinical, functional, and radiological evolution after 24 months of follow‐up of a patient diagnosis of aPAP refractory to WLL, with performed three therapeutic WLLs spaced 16 and 36 months and serious potentially fatal complications in the last one. RESULTS AND DISSCUSION: After 24 months, no adverse effects have appeared and the great clinical, functional and radiological response is maintained. The patient has been successfully treated with inhaled recombinant human GM‐CSF sargramostim. John Wiley and Sons Inc. 2023-06-10 /pmc/articles/PMC10542988/ /pubmed/37300395 http://dx.doi.org/10.1111/crj.13650 Text en © 2023 The Authors. The Clinical Respiratory Journal published by John Wiley & Sons Ltd. https://creativecommons.org/licenses/by/4.0/This is an open access article under the terms of the http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.
spellingShingle Brief Reports
Oterino‐Moreira, Iván
Linares‐Asensio, María‐Jesús
Sanz‐Márquez, Sira
Pérez‐Encinas, Montserrat
Response to inhaled granulocyte‐macrophage colony‐stimulating factor in patient with mild‐to‐moderate autoimmune pulmonary alveolar proteinosis—24 months of follow‐up
title Response to inhaled granulocyte‐macrophage colony‐stimulating factor in patient with mild‐to‐moderate autoimmune pulmonary alveolar proteinosis—24 months of follow‐up
title_full Response to inhaled granulocyte‐macrophage colony‐stimulating factor in patient with mild‐to‐moderate autoimmune pulmonary alveolar proteinosis—24 months of follow‐up
title_fullStr Response to inhaled granulocyte‐macrophage colony‐stimulating factor in patient with mild‐to‐moderate autoimmune pulmonary alveolar proteinosis—24 months of follow‐up
title_full_unstemmed Response to inhaled granulocyte‐macrophage colony‐stimulating factor in patient with mild‐to‐moderate autoimmune pulmonary alveolar proteinosis—24 months of follow‐up
title_short Response to inhaled granulocyte‐macrophage colony‐stimulating factor in patient with mild‐to‐moderate autoimmune pulmonary alveolar proteinosis—24 months of follow‐up
title_sort response to inhaled granulocyte‐macrophage colony‐stimulating factor in patient with mild‐to‐moderate autoimmune pulmonary alveolar proteinosis—24 months of follow‐up
topic Brief Reports
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10542988/
https://www.ncbi.nlm.nih.gov/pubmed/37300395
http://dx.doi.org/10.1111/crj.13650
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