Evidence map›Paper›PMID 38825644›Full record

ReviewExperimental & molecular medicine2024

Advances and challenges in modeling inherited peripheral neuropathies using iPSCs.

Jonas Van Lent, Robert Prior, Gonzalo Pérez Siles, Anthony N Cutrupi, Marina L Kennerson, Tim Vangansewinkel, Esther Wolfs, Bipasha Mukherjee-Clavin, Zachary Nevin, Luke Judge and 7 more

Abstract readReview
In one paragraph

Review in Experimental & molecular medicine, 2024. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 11 papers.

0numbers the graph read from it
0cells of the map it votes in
11citing 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

11 citing papers in PubMed.

  1. Review
  2. Junctions in Jeopardy: the neuromuscular junction is a selective pathological target in Charcot-Marie-Tooth disease.Mammalian genome : official journal of the International Mammalian Genome Society · 2026
    Review
  3. Article
  4. Charcot-Marie-Tooth disease and related neuropathies.Nature reviews. Disease primers · 2026
    Review
  5. Review
  6. Review
  7. Review
  8. Article
  9. Review
  10. Review
  11. Review
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

17 authors.

Jonas Van LentPeripheral Neuropathy Research Group, Department of Biomedical Sciences, University of Antwerp, 2610, Antwerp, Belgium.
Robert PriorUniversitätsklinikum Bonn (UKB), University of Bonn, Bonn, Germany.
Gonzalo Pérez SilesNorthcott Neuroscience Laboratory, ANZAC Research Institute Sydney Local Health District and Faculty of Medicine and Health, University of Sydney, Sydney, NSW, Australia.
Anthony N CutrupiNorthcott Neuroscience Laboratory, ANZAC Research Institute Sydney Local Health District and Faculty of Medicine and Health, University of Sydney, Sydney, NSW, Australia.
Marina L KennersonNorthcott Neuroscience Laboratory, ANZAC Research Institute Sydney Local Health District and Faculty of Medicine and Health, University of Sydney, Sydney, NSW, Australia.
Tim VangansewinkelUHasselt - Hasselt University, BIOMED, Laboratory for Functional Imaging and Research on Stem Cells (FIERCE Lab), Agoralaan, 3590, Diepenbeek, Belgium.
Esther WolfsUHasselt - Hasselt University, BIOMED, Laboratory for Functional Imaging and Research on Stem Cells (FIERCE Lab), Agoralaan, 3590, Diepenbeek, Belgium.
Bipasha Mukherjee-ClavinDepartment of Neurology, Johns Hopkins University School of Medicine, Baltimore, MD, USA.
Zachary NevinGladstone Institutes, San Francisco, CA, USA.ORCID http://orcid.org/0000-0003-4987-924X
Luke JudgeGladstone Institutes, San Francisco, CA, USA.
Bruce ConklinGladstone Institutes, San Francisco, CA, USA.ORCID http://orcid.org/0000-0003-1463-6061
Henna TyynismaaStem Cells and Metabolism Research Program, Faculty of Medicine, University of Helsinki, 00290, Helsinki, Finland.
Alex J ClarkBlizard Institute, Barts and the London School of Medicine and Dentistry, Queen Mary University of London, London, UK.
David L BennettNuffield Department of Clinical Neuroscience, Oxford University, Oxford, UK.ORCID http://orcid.org/0000-0002-7996-2696
Ludo Van Den BoschVIB-Center for Brain and Disease Research, Laboratory of Neurobiology, 3000, Leuven, Belgium.ORCID http://orcid.org/0000-0003-0104-4067
Mario SaportaDepartment of Neurology, Miller School of Medicine, University of Miami, Miami, FL, USA.
Vincent TimmermanPeripheral Neuropathy Research Group, Department of Biomedical Sciences, University of Antwerp, 2610, Antwerp, Belgium. vincent.timmerman@uantwerpen.be.ORCID http://orcid.org/0000-0002-2162-0933

Funding

C9orf72 frontotemporal dementia (FTD) and amyotrophic lateral sclerosis(ALS): using patient cells and CRISPR to reveal therapeutic approachesRF1AG072052 · NIA · J. DAVID GLADSTONE INSTITUTES · PI CONKLIN, BRUCE R · 2021 to 2022
$2.4M
JAX-Gladstone, SCGE Disease Models Studies SupplementU01ES032673 · NIEHS · J. DAVID GLADSTONE INSTITUTES · PI CONKLIN, BRUCE R · 2020 to 2022
$2.4M
Allele-specific inactivation for dominant negative NEFL MutationsR01NS119678 · NINDS · UNIVERSITY OF CALIFORNIA, SAN FRANCISCO · PI JUDGE, LUKE M · 2021 to 2025
$1.9M
C9orf72 frontotemporal dementia (FTD) and amyotrophic lateral sclerosis(ALS): using patient cells and CRISPR to reveal therapeutic approachesR01AG072052 · NIA · J. DAVID GLADSTONE INSTITUTES · PI CONKLIN, BRUCE R · 2024 to 2025
$1.4M
NIA NIH HHS R01 AG072052NIA NIH HHS RF1 AG072052NIEHS NIH HHS U01 ES032673NINDS NIH HHS R01 NS119678Universiteit Antwerpen (University of Antwerp) DOC-PRO4Wellcome Trust
6 · The paper itself

Abstract

Inherited peripheral neuropathies (IPNs) are a group of diseases associated with mutations in various genes with fundamental roles in the development and function of peripheral nerves. Over the past 10 years, significant advances in identifying molecular disease mechanisms underlying axonal and myelin degeneration, acquired from cellular biology studies and transgenic fly and rodent models, have facilitated the development of promising treatment strategies. However, no clinical treatment has emerged to date. This lack of treatment highlights the urgent need for more biologically and clinically relevant models recapitulating IPNs. For both neurodevelopmental and neurodegenerative diseases, patient-specific induced pluripotent stem cells (iPSCs) are a particularly powerful platform for disease modeling and preclinical studies. In this review, we provide an update on different in vitro human cellular IPN models, including traditional two-dimensional monoculture iPSC derivatives, and recent advances in more complex human iPSC-based systems using microfluidic chips, organoids, and assembloids.

Indexed as

Induced Pluripotent Stem CellsAnimalsHumansModels, BiologicalOrganoidsPeripheral Nervous System Diseases

Identifiers

PMID38825644
PMCPMC11263568

What OpenQuestion holds

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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.