Evidence map›Paper›PMID 33945785›Full record

ArticleDevelopmental cell2021

iPSC-derived myelinoids to study myelin biology of humans.

Owen G James, Bhuvaneish T Selvaraj, Dario Magnani, Karen Burr, Peter Connick, Samantha K Barton, Navneet A Vasistha, David W Hampton, David Story, Robert Smigiel and 5 more

Erratum issuedOpen access · hybridAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Developmental cell, 2021. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 58 papers.

0numbers the graph read from it
0cells of the map it votes in
58citing papers in PubMed
7.7field-weighted citation impact, top 1% 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

58 citing papers in PubMed, 81 citations in OpenAlex.

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4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

15 authors at 5 institutions in 4 countries.

Owen G JamesUK Dementia Research Institute at the University of Edinburgh, Edinburgh EH16 4SB, UK; Centre for Clinical Brain Sciences, University of Edinburgh, Edinburgh EH16 4SB, UK; Euan MacDonald Centre for Motor Neurone Disease Research University of Edinburgh, Edinburgh EH16 4SB, UK; Anne Rowling Regenerative Neurology Clinic, University of Edinburgh, Edinburgh EH16 4SB, UK.
Bhuvaneish T SelvarajUK Dementia Research Institute at the University of Edinburgh, Edinburgh EH16 4SB, UK; Centre for Clinical Brain Sciences, University of Edinburgh, Edinburgh EH16 4SB, UK; Euan MacDonald Centre for Motor Neurone Disease Research University of Edinburgh, Edinburgh EH16 4SB, UK.
Dario MagnaniUK Dementia Research Institute at the University of Edinburgh, Edinburgh EH16 4SB, UK; Centre for Clinical Brain Sciences, University of Edinburgh, Edinburgh EH16 4SB, UK; Euan MacDonald Centre for Motor Neurone Disease Research University of Edinburgh, Edinburgh EH16 4SB, UK.
Karen BurrUK Dementia Research Institute at the University of Edinburgh, Edinburgh EH16 4SB, UK; Centre for Clinical Brain Sciences, University of Edinburgh, Edinburgh EH16 4SB, UK; Euan MacDonald Centre for Motor Neurone Disease Research University of Edinburgh, Edinburgh EH16 4SB, UK.
Peter ConnickUK Dementia Research Institute at the University of Edinburgh, Edinburgh EH16 4SB, UK; Centre for Clinical Brain Sciences, University of Edinburgh, Edinburgh EH16 4SB, UK; Anne Rowling Regenerative Neurology Clinic, University of Edinburgh, Edinburgh EH16 4SB, UK.
Samantha K BartonUK Dementia Research Institute at the University of Edinburgh, Edinburgh EH16 4SB, UK; Centre for Clinical Brain Sciences, University of Edinburgh, Edinburgh EH16 4SB, UK; Euan MacDonald Centre for Motor Neurone Disease Research University of Edinburgh, Edinburgh EH16 4SB, UK; Florey Institute of Neuroscience and Mental Health, University of Melbourne, Melbourne, Australia.
Navneet A VasisthaCentre for Clinical Brain Sciences, University of Edinburgh, Edinburgh EH16 4SB, UK; Euan MacDonald Centre for Motor Neurone Disease Research University of Edinburgh, Edinburgh EH16 4SB, UK; Biotech Research and Innovation Centre, Copenhagen N 2200, Denmark.
David W HamptonUK Dementia Research Institute at the University of Edinburgh, Edinburgh EH16 4SB, UK; Centre for Clinical Brain Sciences, University of Edinburgh, Edinburgh EH16 4SB, UK; Euan MacDonald Centre for Motor Neurone Disease Research University of Edinburgh, Edinburgh EH16 4SB, UK.
David StoryUK Dementia Research Institute at the University of Edinburgh, Edinburgh EH16 4SB, UK; Centre for Clinical Brain Sciences, University of Edinburgh, Edinburgh EH16 4SB, UK; Euan MacDonald Centre for Motor Neurone Disease Research University of Edinburgh, Edinburgh EH16 4SB, UK.
Robert SmigielDepartment of Pediatrics and Rare Disorders, Wroclaw Medical University, Wrocław 51-618, Poland.
Rafal PloskiDepartment of Medical Genetics, Medical University of Warsaw, Warsaw 02-106, Poland.
Peter J BrophyCentre for Discovery Brain Sciences, University of Edinburgh, Edinburgh EH16 4SB, UK.
Charles Ffrench-ConstantMRC Centre for Regenerative Medicine, University of Edinburgh, Edinburgh EH16 4UU, UK.
David A LyonsCentre for Discovery Brain Sciences, University of Edinburgh, Edinburgh EH16 4SB, UK.
Siddharthan ChandranUK Dementia Research Institute at the University of Edinburgh, Edinburgh EH16 4SB, UK; Centre for Clinical Brain Sciences, University of Edinburgh, Edinburgh EH16 4SB, UK; Euan MacDonald Centre for Motor Neurone Disease Research University of Edinburgh, Edinburgh EH16 4SB, UK; Anne Rowling Regenerative Neurology Clinic, University of Edinburgh, Edinburgh EH16 4SB, UK; Centre for Brain Development and Repair, inStem, Bangalore 560065, India. Electronic address: siddharthan.chandran@ed.ac.uk.
University of Edinburgh · GBMott MacDonald (United Kingdom) · GBMedical University of Warsaw · PLMRC Centre for Regenerative Medicine · GBWroclaw Medical University · PL

Funding

Medical Research Council G0601744Medical Research Council MR/L023784/2Medical Research Council MR/N013255/1Medical Research Council MR/P006272/1Wellcome TrustWellcome Trust 102836/Z/13/ZWellcome Trust 107008Wellcome Trust 214244/Z/18/Z
6 · The paper itself

Abstract

Myelination is essential for central nervous system (CNS) formation, health, and function. Emerging evidence of oligodendrocyte heterogeneity in health and disease and divergent CNS gene expression profiles between mice and humans supports the development of experimentally tractable human myelination systems. Here, we developed human iPSC-derived myelinating organoids ("myelinoids") and quantitative tools to study myelination from oligodendrogenesis through to compact myelin formation and myelinated axon organization. Using patient-derived cells, we modeled a monogenetic disease of myelinated axons (Nfasc155 deficiency), recapitulating impaired paranodal axo-glial junction formation. We also validated the use of myelinoids for pharmacological assessment of myelination-both at the level of individual oligodendrocytes and globally across whole myelinoids-and demonstrated reduced myelination in response to suppressed synaptic vesicle release. Our study provides a platform to investigate human myelin development, disease, and adaptive myelination.

Indexed as

AxonsHumansInduced Pluripotent Stem CellsMyelin SheathNerve Growth FactorsOrganoidsTetanus ToxinTime FactorsNerve Growth FactorsTetanus Toxinadaptive myelinationdisease modellinghuman myelinationhuman stem celliPSCmyelinmyelinoidoligodendrocyteorganoidparanode

Identifiers

PMID33945785
PMCPMC8098746
OpenAlexW3158405122

What OpenQuestion holds

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Registered trials

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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.