Pulmonary Alveolar Proteinosis Refractory to Plasmapheresis and Rituximab despite GM-CSF Antibody Reduction

We share our experience of a patient with pulmonary alveolar proteinosis who was refractory to plasmapheresis and rituximab despite a significant reduction in the offending antibody. He presented with shortness of breath, fevers, chills, and sweats for 4 months. He was diagnosed with autoimmune PAP...

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Main Authors: Aysenur Keske, Eric M. Destrampe, Byron Barksdale, William N. Rose
Format: Article
Language:English
Published: Wiley 2022-01-01
Series:Case Reports in Immunology
Online Access:http://dx.doi.org/10.1155/2022/2104270
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author Aysenur Keske
Eric M. Destrampe
Byron Barksdale
William N. Rose
author_facet Aysenur Keske
Eric M. Destrampe
Byron Barksdale
William N. Rose
author_sort Aysenur Keske
collection DOAJ
description We share our experience of a patient with pulmonary alveolar proteinosis who was refractory to plasmapheresis and rituximab despite a significant reduction in the offending antibody. He presented with shortness of breath, fevers, chills, and sweats for 4 months. He was diagnosed with autoimmune PAP based on typical radiology findings, bronchoalveolar fluid analysis, and elevated anti-GM-CSF levels. Given his limited improvement with whole lung lavage and inhaled GM-CSF therapy, he underwent two series of plasmapheresis. Series one was 5 procedures in 6 days, and series two was 5 procedures in 9 days followed by rituximab. These did not appear to provide any lasting clinical benefit in the year after plasmapheresis despite a marked decrease in serum anti-GM-CSF levels. However, about a year after plasmapheresis, he went into remission and has not required any treatment.
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spelling doaj-art-4aef6cb4317b40f49debc19a0da5eba72025-08-20T03:36:26ZengWileyCase Reports in Immunology2090-66172022-01-01202210.1155/2022/2104270Pulmonary Alveolar Proteinosis Refractory to Plasmapheresis and Rituximab despite GM-CSF Antibody ReductionAysenur Keske0Eric M. Destrampe1Byron Barksdale2William N. Rose3Department of PathologyDepartment of PathologyDepartment of PathologyDepartment of PathologyWe share our experience of a patient with pulmonary alveolar proteinosis who was refractory to plasmapheresis and rituximab despite a significant reduction in the offending antibody. He presented with shortness of breath, fevers, chills, and sweats for 4 months. He was diagnosed with autoimmune PAP based on typical radiology findings, bronchoalveolar fluid analysis, and elevated anti-GM-CSF levels. Given his limited improvement with whole lung lavage and inhaled GM-CSF therapy, he underwent two series of plasmapheresis. Series one was 5 procedures in 6 days, and series two was 5 procedures in 9 days followed by rituximab. These did not appear to provide any lasting clinical benefit in the year after plasmapheresis despite a marked decrease in serum anti-GM-CSF levels. However, about a year after plasmapheresis, he went into remission and has not required any treatment.http://dx.doi.org/10.1155/2022/2104270
spellingShingle Aysenur Keske
Eric M. Destrampe
Byron Barksdale
William N. Rose
Pulmonary Alveolar Proteinosis Refractory to Plasmapheresis and Rituximab despite GM-CSF Antibody Reduction
Case Reports in Immunology
title Pulmonary Alveolar Proteinosis Refractory to Plasmapheresis and Rituximab despite GM-CSF Antibody Reduction
title_full Pulmonary Alveolar Proteinosis Refractory to Plasmapheresis and Rituximab despite GM-CSF Antibody Reduction
title_fullStr Pulmonary Alveolar Proteinosis Refractory to Plasmapheresis and Rituximab despite GM-CSF Antibody Reduction
title_full_unstemmed Pulmonary Alveolar Proteinosis Refractory to Plasmapheresis and Rituximab despite GM-CSF Antibody Reduction
title_short Pulmonary Alveolar Proteinosis Refractory to Plasmapheresis and Rituximab despite GM-CSF Antibody Reduction
title_sort pulmonary alveolar proteinosis refractory to plasmapheresis and rituximab despite gm csf antibody reduction
url http://dx.doi.org/10.1155/2022/2104270
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AT byronbarksdale pulmonaryalveolarproteinosisrefractorytoplasmapheresisandrituximabdespitegmcsfantibodyreduction
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