Evidence map›Paper›PMID 38114482›Full record

ArticleNature communications2023

Efficient generation of a self-organizing neuromuscular junction model from human pluripotent stem cells.

Alessia Urzi, Ines Lahmann, Lan Vi N Nguyen, Benjamin R Rost, Angélica García-Pérez, Noemie Lelievre, Megan E Merritt-Garza, Han C Phan, Gary J Bassell, Wilfried Rossoll and 4 more

Abstract read
In one paragraph

Article in Nature communications, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 17 papers.

0numbers the graph read from it
0cells of the map it votes in
17citing papers in PubMed
–field-weighted citation impact
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

17 citing papers in PubMed.

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  16. Aberrant evoked calcium signaling and nAChR cluster morphology in aFrontiers in cell and developmental biology · 2024
    Article
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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.

Alessia UrziStem Cell Modeling of Development & Disease Group, Max Delbrück Center for Molecular Medicine in the Helmholtz Association (MDC), 13125, Berlin, Germany.
Ines Lahmann *Stem Cell Modeling of Development & Disease Group, Max Delbrück Center for Molecular Medicine in the Helmholtz Association (MDC), 13125, Berlin, Germany.
Lan Vi N Nguyen *Stem Cell Modeling of Development & Disease Group, Max Delbrück Center for Molecular Medicine in the Helmholtz Association (MDC), 13125, Berlin, Germany.
Benjamin R Rost *German Center for Neurodegenerative Diseases (DZNE), Berlin, Germany.ORCID 0000-0003-1906-0081
Angélica García-PérezStem Cell Modeling of Development & Disease Group, Max Delbrück Center for Molecular Medicine in the Helmholtz Association (MDC), 13125, Berlin, Germany.
Noemie LelievreStem Cell Modeling of Development & Disease Group, Max Delbrück Center for Molecular Medicine in the Helmholtz Association (MDC), 13125, Berlin, Germany.
Megan E Merritt-GarzaDepartment of Cell Biology, Laboratory for Translational Cell Biology, Emory University School of Medicine, Atlanta, GA, 30322, USA.
Han C PhanDepartment of Pediatrics, University of Alabama, Birmingham, AL, 35294, USA.
Gary J BassellDepartment of Cell Biology, Laboratory for Translational Cell Biology, Emory University School of Medicine, Atlanta, GA, 30322, USA.ORCID 0000-0003-2622-0127
Wilfried RossollDepartment of Neuroscience, Mayo Clinic, Jacksonville, FL, 32224, USA.ORCID 0000-0002-9622-541X
Sebastian DieckeMax Delbrück Center for Molecular Medicine in the Helmholtz Association (MDC), Technology Platform Pluripotent Stem Cells, 13125, Berlin, Germany.ORCID 0000-0002-5219-5992
Severine KunzMax Delbrück Center for Molecular Medicine in the Helmholtz Association (MDC), Technology Platform Electron Microscopy, 13125, Berlin, Germany.ORCID 0000-0002-0131-3506
Dietmar SchmitzGerman Center for Neurodegenerative Diseases (DZNE), Berlin, Germany.
Mina GoutiStem Cell Modeling of Development & Disease Group, Max Delbrück Center for Molecular Medicine in the Helmholtz Association (MDC), 13125, Berlin, Germany. mina.gouti@mdc-berlin.de.ORCID 0000-0001-7422-1605

Funding

RNA Processing Defects in SMA and Their Contribution to the Disease PhenotypeR01NS091749 · NINDS · MAYO CLINIC JACKSONVILLE · PI ROSSOLL, WILFRIED · 2015 to 2019
$1.9M
NINDS NIH HHS R01 NS091749
6 · The paper itself

Abstract

The complex neuromuscular network that controls body movements is the target of severe diseases that result in paralysis and death. Here, we report the development of a robust and efficient self-organizing neuromuscular junction (soNMJ) model from human pluripotent stem cells that can be maintained long-term in simple adherent conditions. The timely application of specific patterning signals instructs the simultaneous development and differentiation of position-specific brachial spinal neurons, skeletal muscles, and terminal Schwann cells. High-content imaging reveals self-organized bundles of aligned muscle fibers surrounded by innervating motor neurons that form functional neuromuscular junctions. Optogenetic activation and pharmacological interventions show that the spinal neurons actively instruct the synchronous skeletal muscle contraction. The generation of a soNMJ model from spinal muscular atrophy patient-specific iPSCs reveals that the number of NMJs and muscle contraction is severely affected, resembling the patient's pathology. In the future, the soNMJ model could be used for high-throughput studies in disease modeling and drug development. Thus, this model will allow us to address unmet needs in the neuromuscular disease field.

Indexed as

Induced Pluripotent Stem CellsMuscular Atrophy, SpinalHumansMotor NeuronsMuscle Fibers, SkeletalMuscle, SkeletalNeuromuscular Junction

Identifiers

PMID38114482
PMCPMC10730704

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.