Evidence map›Paper›PMID 27453007›Full record

ArticleStem cell reports2016

Murine iPSC-Derived Macrophages as a Tool for Disease Modeling of Hereditary Pulmonary Alveolar Proteinosis due to Csf2rb Deficiency.

Adele Mucci, Jessica Kunkiel, Takuji Suzuki, Sebastian Brennig, Silke Glage, Mark P Kühnel, Mania Ackermann, Christine Happle, Alexandra Kuhn, Axel Schambach and 4 more

Open access · goldAbstract read
In one paragraph

Article in Stem cell reports, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 18 papers.

0numbers the graph read from it
0cells of the map it votes in
18citing papers in PubMed
3.8field-weighted citation impact, top 6% 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

18 citing papers in PubMed, 24 citations in OpenAlex.

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  17. Gene correction ofBlood advances · 2017
    Article
  18. Ex vivo Generation of Genetically Modified Macrophages from Human Induced Pluripotent Stem Cells.Transfusion medicine and hemotherapy : offizielles Organ der Deutschen Gesellschaft fur Transfusionsmedizin und Immunhamatologie · 2017
    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

14 authors at 4 institutions in 2 countries.

Adele MucciResearch Group Reprogramming and Gene Therapy, Cluster of Excellence REBIRTH, Hannover Medical School, 30625 Hannover, Germany; Junior Research Group Translational Hematology of Congenital Diseases, Cluster of Excellence REBIRTH, Hannover Medical School, Carl-Neuberg-Street 1, 30625 Hannover, Germany; Institute of Experimental Hematology, Hannover Medical School, 30625 Hannover, Germany.
Jessica KunkielResearch Group Reprogramming and Gene Therapy, Cluster of Excellence REBIRTH, Hannover Medical School, 30625 Hannover, Germany; Institute of Experimental Hematology, Hannover Medical School, 30625 Hannover, Germany.
Takuji SuzukiTranslational Pulmonary Science Center, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA.
Sebastian BrennigResearch Group Reprogramming and Gene Therapy, Cluster of Excellence REBIRTH, Hannover Medical School, 30625 Hannover, Germany; Junior Research Group Translational Hematology of Congenital Diseases, Cluster of Excellence REBIRTH, Hannover Medical School, Carl-Neuberg-Street 1, 30625 Hannover, Germany; Institute of Experimental Hematology, Hannover Medical School, 30625 Hannover, Germany.
Silke GlageInstitute of Laboratory Animal Science and Central Animal Facility, Hannover Medical School, 30625 Hannover, Germany.
Mark P KühnelDepartment of Functional and Applied Anatomy, Hannover Medical School, 30625 Hannover, Germany.
Mania AckermannResearch Group Reprogramming and Gene Therapy, Cluster of Excellence REBIRTH, Hannover Medical School, 30625 Hannover, Germany; Junior Research Group Translational Hematology of Congenital Diseases, Cluster of Excellence REBIRTH, Hannover Medical School, Carl-Neuberg-Street 1, 30625 Hannover, Germany; Institute of Experimental Hematology, Hannover Medical School, 30625 Hannover, Germany.
Christine HappleDepartment of Pediatric Pneumology, Allergology and Neonatology, Hannover Medical School, 30625 Hannover, Germany; Biomedical Research in Endstage and Obstructive Lung Disease Hannover (BREATH), Member of the German Center for Lung Research (DZL), 30625 Hannover, Germany.
Alexandra KuhnResearch Group Reprogramming and Gene Therapy, Cluster of Excellence REBIRTH, Hannover Medical School, 30625 Hannover, Germany; Institute of Experimental Hematology, Hannover Medical School, 30625 Hannover, Germany.
Axel SchambachInstitute of Experimental Hematology, Hannover Medical School, 30625 Hannover, Germany; Division of Hematology/Oncology, Boston Children's Hospital, Harvard Medical School, Boston, MA 02115, USA.
Bruce C TrapnellTranslational Pulmonary Science Center, Cincinnati Children's Hospital Medical Center, Cincinnati, OH 45229, USA.
Gesine HansenDepartment of Pediatric Pneumology, Allergology and Neonatology, Hannover Medical School, 30625 Hannover, Germany; Biomedical Research in Endstage and Obstructive Lung Disease Hannover (BREATH), Member of the German Center for Lung Research (DZL), 30625 Hannover, Germany.
Thomas MoritzResearch Group Reprogramming and Gene Therapy, Cluster of Excellence REBIRTH, Hannover Medical School, 30625 Hannover, Germany; Institute of Experimental Hematology, Hannover Medical School, 30625 Hannover, Germany.
Nico LachmannJunior Research Group Translational Hematology of Congenital Diseases, Cluster of Excellence REBIRTH, Hannover Medical School, Carl-Neuberg-Street 1, 30625 Hannover, Germany; Institute of Experimental Hematology, Hannover Medical School, 30625 Hannover, Germany. Electronic address: lachmann.nico@mh-hannover.de.
Medizinische Hochschule Hannover · DECincinnati Children's Hospital Medical Center · USGerman Center for Lung Research · DEBoston Children's Hospital · US

Funding

LCenter for Clinical and Translational Science and TrainingUL1TR001425 · NCATS · UNIVERSITY OF CINCINNATI · PI HEUBI, JAMES E., KISSELA, BRETT M · 2015 to 2024
$37.5M
Role of GM-CSF in Myeloid Cell Function and Innate ImmunityR01HL085453 · NHLBI · CINCINNATI CHILDRENS HOSP MED CTR · PI TRAPNELL, BRUCE C · 2007 to 2023
$6.5M
Pulmonary Macrophage Transplantation for Pulmonary Alveolar ProteinosisR01HL118342 · NHLBI · CINCINNATI CHILDRENS HOSP MED CTR · PI TRAPNELL, BRUCE C, TRAPNELL, BRUCE COLSTON · 2014 to 2021
$4.5M
NCATS NIH HHS UL1 TR001425NHLBI NIH HHS R01 HL085453NHLBI NIH HHS R01 HL118342
6 · The paper itself

Abstract

Induced pluripotent stem cells (iPSCs) represent an innovative source for the standardized in vitro generation of macrophages (Mφ). We here describe a robust and efficient protocol to obtain mature and functional Mφ from healthy as well as disease-specific murine iPSCs. With regard to morphology, surface phenotype, and function, our iPSC-derived Mφ (iPSC-Mφ) closely resemble their counterparts generated in vitro from bone marrow cells. Moreover, when we investigated the feasibility of our differentiation system to serve as a model for rare congenital diseases associated with Mφ malfunction, we were able to faithfully recapitulate the pathognomonic defects in GM-CSF signaling and Mφ function present in hereditary pulmonary alveolar proteinosis (herPAP). Thus, our studies may help to overcome the limitations placed on research into certain rare disease entities by the lack of an adequate supply of disease-specific primary cells, and may aid the development of novel therapeutic approaches for herPAP patients.

Indexed as

AnimalsBiomarkersCell DifferentiationCytokine Receptor Common beta SubunitDisease Models, AnimalGranulocyte-Macrophage Colony-Stimulating FactorHematopoiesisInduced Pluripotent Stem CellsMacrophagesMice, Inbred C57BLMonocytesPulmonary Alveolar ProteinosisBiomarkersCsf2rb protein, mouseCytokine Receptor Common beta SubunitGranulocyte-Macrophage Colony-Stimulating Factordisease modelinghematopoiesisiPSCmacrophagesPAP

Identifiers

PMID27453007
PMCPMC4982988
OpenAlexW2506000063

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
Read underepoch 390

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.