Evidence map›Paper›PMID 35928826›Full record

ArticleFrontiers in immunology2022

Allogeneic Hematopoietic Stem Cell Transplantation After Prior Lung Transplantation for Hereditary Pulmonary Alveolar Proteinosis: A Case Report.

Hanne Beeckmans, Gene P L Ambrocio, Saskia Bos, Astrid Vermaut, Vincent Geudens, Arno Vanstapel, Bart M Vanaudenaerde, Frans De Baets, Thomas L A Malfait, Marie-Paule Emonds and 3 more

Open access · goldAbstract readCase Reports
In one paragraph

Article in Frontiers in immunology, 2022. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 10 papers.

0numbers the graph read from it
0cells of the map it votes in
10citing papers in PubMed
2.3field-weighted citation impact, top 11% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

10 citing papers in PubMed, 15 citations in OpenAlex.

  1. Article
  2. The dark side of pulmonary alveolar proteinosis.Multidisciplinary respiratory medicine · 2025
    Article
  3. Review
  4. Article
  5. Article
  6. Pathogenesis-driven treatment of primary pulmonary alveolar proteinosis.European respiratory review : an official journal of the European Respiratory Society · 2024
    Review
  7. A toxicology study ofMolecular therapy. Methods & clinical development · 2024
    Article
  8. Article
  9. ERJ open research · 2023
    Article
  10. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

13 authors at 4 institutions in 3 countries.

Hanne BeeckmansDepartment of Department of Chronic Diseases and Metabolism (CHROMETA), Laboratory of Respiratory Diseases and Thoracic Surgery (BREATHE), KU Leuven, Leuven, Belgium.
Gene P L AmbrocioDepartment of Internal Medicine, Division of Pulmonary Medicine, University of the Philippines - Philippine General Hospital, Manila, Philippines.
Saskia BosTranslational and Clinical Research Institute, Newcastle University, Newcastle Upon Tyne, United Kingdom.
Astrid VermautDepartment of Department of Chronic Diseases and Metabolism (CHROMETA), Laboratory of Respiratory Diseases and Thoracic Surgery (BREATHE), KU Leuven, Leuven, Belgium.
Vincent GeudensDepartment of Department of Chronic Diseases and Metabolism (CHROMETA), Laboratory of Respiratory Diseases and Thoracic Surgery (BREATHE), KU Leuven, Leuven, Belgium.
Arno VanstapelDepartment of Department of Chronic Diseases and Metabolism (CHROMETA), Laboratory of Respiratory Diseases and Thoracic Surgery (BREATHE), KU Leuven, Leuven, Belgium.
Bart M VanaudenaerdeDepartment of Department of Chronic Diseases and Metabolism (CHROMETA), Laboratory of Respiratory Diseases and Thoracic Surgery (BREATHE), KU Leuven, Leuven, Belgium.
Frans De BaetsDepartment of Pediatrics, Ghent University Hospital, Ghent, Belgium.
Thomas L A MalfaitDepartment of Respiratory Medicine, Ghent University Hospital, Ghent, Belgium.
Marie-Paule EmondsHistocompatibility and Immunogenetics Laboratory, Red Cross-Flanders, Mechelen, Belgium.
Dirk E Van RaemdonckDepartment of Department of Chronic Diseases and Metabolism (CHROMETA), Laboratory of Respiratory Diseases and Thoracic Surgery (BREATHE), KU Leuven, Leuven, Belgium.
Hélène M SchoemansDepartment of Hematology, Bone Marrow Transplant Unit, University Hospitals Leuven, Leuven, Belgium.
Robin VosDepartment of Department of Chronic Diseases and Metabolism (CHROMETA), Laboratory of Respiratory Diseases and Thoracic Surgery (BREATHE), KU Leuven, Leuven, Belgium.
KU Leuven · BEGhent University Hospital · BENewcastle University · GBPhilippine General Hospital · PH

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Pulmonary alveolar proteinosis (PAP) is a rare, diffuse lung disorder characterized by surfactant accumulation in the small airways due to defective clearance by alveolar macrophages, resulting in impaired gas exchange. Whole lung lavage is the current standard of care treatment for PAP. Lung transplantation is an accepted treatment option when whole lung lavage or other experimental treatment options are ineffective, or in case of extensive pulmonary fibrosis secondary to PAP. A disadvantage of lung transplantation is recurrence of PAP in the transplanted lungs, especially in hereditary PAP. The hereditary form of PAP is an ultra-rare condition caused by genetic mutations in genes encoding for the granulocyte macrophage-colony stimulating factor (GM-CSF) receptor, and intrinsically affects bone marrow derived-monocytes, which differentiate into macrophages in the lung. Consequently, these macrophages typically display disrupted GM-CSF receptor-signaling, causing defective surfactant clearance. Bone marrow/hematopoietic stem cell transplantation may potentially reverse the lung disease in hereditary PAP. In patients with hereditary PAP undergoing lung transplantation, post-lung transplant recurrence of PAP may theoretically be averted by subsequent hematopoietic stem cell transplantation, which results in a graft-versus-

Indexed as

Hematopoietic Stem Cell TransplantationLung TransplantationPulmonary Alveolar ProteinosisPulmonary FibrosisPulmonary SurfactantsHumansQuality of LifeReceptors, Granulocyte-Macrophage Colony-Stimulating FactorSurface-Active AgentsPulmonary SurfactantsReceptors, Granulocyte-Macrophage Colony-Stimulating FactorSurface-Active Agentsallogeneic hematopoietic stem cell transplantationbone marrow transplantationlung transplantationpulmonary alveolar proteinosisrejection

Identifiers

PMID35928826
PMCPMC9344132
OpenAlexW4286239302

What OpenQuestion holds

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LicenceCC BY
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Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.