Pulmonary alveolar proteinosis after lung transplantation: Two case reports and literature review

Abstract Pulmonary alveolar proteinosis (PAP) affecting transplanted lungs is not well recognized. Herein, we report two cases of PAP after lung transplantation (LTx). The first case was a 4‐year‐old boy with hereditary pulmonary fibrosis who underwent bilateral LTx and presented with respiratory di...

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Main Authors: Shinichi Kawana, Kentaroh Miyoshi, Shin Tanaka, Seiichiro Sugimoto, Dai Shimizu, Kei Matsubara, Mikio Okazaki, Noboru Hattori, Shinichi Toyooka
Format: Article
Language:English
Published: Wiley 2023-06-01
Series:Respirology Case Reports
Subjects:
Online Access:https://doi.org/10.1002/rcr2.1160
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author Shinichi Kawana
Kentaroh Miyoshi
Shin Tanaka
Seiichiro Sugimoto
Dai Shimizu
Kei Matsubara
Mikio Okazaki
Noboru Hattori
Shinichi Toyooka
author_facet Shinichi Kawana
Kentaroh Miyoshi
Shin Tanaka
Seiichiro Sugimoto
Dai Shimizu
Kei Matsubara
Mikio Okazaki
Noboru Hattori
Shinichi Toyooka
author_sort Shinichi Kawana
collection DOAJ
description Abstract Pulmonary alveolar proteinosis (PAP) affecting transplanted lungs is not well recognized. Herein, we report two cases of PAP after lung transplantation (LTx). The first case was a 4‐year‐old boy with hereditary pulmonary fibrosis who underwent bilateral LTx and presented with respiratory distress on postoperative day (POD) 23. He was initially treated for acute rejection, died due to infection on POD 248, and was diagnosed with PAP at autopsy. The second case involved a 52‐year‐old man with idiopathic pulmonary fibrosis who underwent bilateral LTx. On POD 99, chest computed tomography revealed ground‐glass opacities. Bronchoalveolar lavage and transbronchial biopsy led to a diagnosis of PAP. Follow‐up with immunosuppression tapering resulted in clinical and radiological improvement. PAP after lung transplantation mimics common acute rejection; however, is potentially transient or resolved with tapering immunosuppression, as observed in the second case. Transplant physicians should be aware of this rare complication to avoid misconducting immunosuppressive management.
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spelling doaj.art-a47f81b23bc243978b5b47c52665d3212023-05-26T03:14:31ZengWileyRespirology Case Reports2051-33802023-06-01116n/an/a10.1002/rcr2.1160Pulmonary alveolar proteinosis after lung transplantation: Two case reports and literature reviewShinichi Kawana0Kentaroh Miyoshi1Shin Tanaka2Seiichiro Sugimoto3Dai Shimizu4Kei Matsubara5Mikio Okazaki6Noboru Hattori7Shinichi Toyooka8Department of General Thoracic Surgery and Organ Transplant Center Okayama University Hospital Okayama JapanDepartment of General Thoracic Surgery and Organ Transplant Center Okayama University Hospital Okayama JapanDepartment of General Thoracic Surgery and Organ Transplant Center Okayama University Hospital Okayama JapanDepartment of General Thoracic Surgery and Organ Transplant Center Okayama University Hospital Okayama JapanDepartment of General Thoracic Surgery and Organ Transplant Center Okayama University Hospital Okayama JapanDepartment of General Thoracic Surgery and Organ Transplant Center Okayama University Hospital Okayama JapanDepartment of General Thoracic Surgery and Organ Transplant Center Okayama University Hospital Okayama JapanDepartment of Molecular and Internal Medicine Hiroshima University, Graduate School of Biomedical and Health Sciences Hiroshima JapanDepartment of General Thoracic Surgery and Organ Transplant Center Okayama University Hospital Okayama JapanAbstract Pulmonary alveolar proteinosis (PAP) affecting transplanted lungs is not well recognized. Herein, we report two cases of PAP after lung transplantation (LTx). The first case was a 4‐year‐old boy with hereditary pulmonary fibrosis who underwent bilateral LTx and presented with respiratory distress on postoperative day (POD) 23. He was initially treated for acute rejection, died due to infection on POD 248, and was diagnosed with PAP at autopsy. The second case involved a 52‐year‐old man with idiopathic pulmonary fibrosis who underwent bilateral LTx. On POD 99, chest computed tomography revealed ground‐glass opacities. Bronchoalveolar lavage and transbronchial biopsy led to a diagnosis of PAP. Follow‐up with immunosuppression tapering resulted in clinical and radiological improvement. PAP after lung transplantation mimics common acute rejection; however, is potentially transient or resolved with tapering immunosuppression, as observed in the second case. Transplant physicians should be aware of this rare complication to avoid misconducting immunosuppressive management.https://doi.org/10.1002/rcr2.1160graft dysfunctionimmunosuppressionlung transplantationpulmonary alveolar proteinosis
spellingShingle Shinichi Kawana
Kentaroh Miyoshi
Shin Tanaka
Seiichiro Sugimoto
Dai Shimizu
Kei Matsubara
Mikio Okazaki
Noboru Hattori
Shinichi Toyooka
Pulmonary alveolar proteinosis after lung transplantation: Two case reports and literature review
Respirology Case Reports
graft dysfunction
immunosuppression
lung transplantation
pulmonary alveolar proteinosis
title Pulmonary alveolar proteinosis after lung transplantation: Two case reports and literature review
title_full Pulmonary alveolar proteinosis after lung transplantation: Two case reports and literature review
title_fullStr Pulmonary alveolar proteinosis after lung transplantation: Two case reports and literature review
title_full_unstemmed Pulmonary alveolar proteinosis after lung transplantation: Two case reports and literature review
title_short Pulmonary alveolar proteinosis after lung transplantation: Two case reports and literature review
title_sort pulmonary alveolar proteinosis after lung transplantation two case reports and literature review
topic graft dysfunction
immunosuppression
lung transplantation
pulmonary alveolar proteinosis
url https://doi.org/10.1002/rcr2.1160
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