ArticleStem cell reports2018
Chemical Screening Identifies Enhancers of Mutant Oligodendrocyte Survival and Unmasks a Distinct Pathological Phase in Pelizaeus-Merzbacher Disease.
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Article in Stem cell reports, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 25 papers.
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25citing papers in PubMed
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Who cites it
25 citing papers in PubMed, 38 citations in OpenAlex.
- Review
- Article
- Article
- Astrocyte and oligodendrocyte pathology in Alzheimer's disease.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2025Review
- Potential Role of Endoplasmic Reticulum Stress in Modulating Protein Homeostasis in Oligodendrocytes to Improve White Matter Injury in Preterm Infants.Molecular neurobiology · 2024Review
- Homozygous EPRS1 missense variant causing hypomyelinating leukodystrophy-15 alters variant-distal mRNA mNature communications · 2024Article
- Pelizaeus-Merzbacher disease: on the cusp of myelin medicine.Trends in molecular medicine · 2024Review
- Alterations in oligodendrocyte transcriptional networks reveal region-specific vulnerabilities to neurological disease.iScience · 2023Article
- High-throughput screening for myelination promoting compounds using human stem cell-derived oligodendrocyte progenitor cells.iScience · 2023Article
- The landscape of targets and lead molecules for remyelination.Nature chemical biology · 2022Review
- Enhancers of Human and Rodent Oligodendrocyte Formation Predominantly Induce Cholesterol Precursor Accumulation.ACS chemical biology · 2022Article
- A multiparametric calcium signal screening platform using iPSC-derived cortical neural spheroids.SLAS discovery : advancing life sciences R & D · 2022Article
- An in vivo accelerated developmental myelination model for testing promyelinating therapeutics.BMC neuroscience · 2022Article
- Genotype-phenotype correlation and natural history analyses in a Chinese cohort with pelizaeus-merzbacher disease.Orphanet journal of rare diseases · 2022Article
- Non-canonical Targets of HIF1a Impair Oligodendrocyte Progenitor Cell Function.Cell stem cell · 2021Article
- Patient-derived iPSC modeling of rare neurodevelopmental disorders: Molecular pathophysiology and prospective therapies.Neuroscience and biobehavioral reviews · 2021Review
- Hypomyelinating leukodystrophies - unravelling myelin biology.Nature reviews. Neurology · 2021Review
- Rapid and Efficient Generation of Myelinating Human Oligodendrocytes in Organoids.Frontiers in cellular neuroscience · 2021Article
- Neuron-Oligodendrocyte Interactions in the Structure and Integrity of Axons.Frontiers in cell and developmental biology · 2021Review
- Electrospun Fiber Scaffolds for Engineering Glial Cell Behavior to Promote Neural Regeneration.Bioengineering (Basel, Switzerland) · 2020Review
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Authors and funding
16 authors at 2 institutions in 1 country.
Matthew S ElittDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
H Elizabeth ShickDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Mayur MadhavanDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Kevin C AllanDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Benjamin L L ClaytonDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Chen WengDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Tyler E MillerDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Daniel C FactorDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Lilianne BarbarDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Baraa S NawashDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Zachary S NevinDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Angela M LagerDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Yan LiDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Fulai JinDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA; Department of Population and Quantitative Health Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA; Department of Engineering and Computer Science, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA; Case Comprehensive Cancer Center, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Drew J AdamsDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA; Case Comprehensive Cancer Center, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Paul J TesarDepartment of Genetics and Genome Sciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA; Department of Neurosciences, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA; Case Comprehensive Cancer Center, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA. Electronic address: paul.tesar@case.edu.
Case Western Reserve University · USUniversity School · US
Funding
TUMOR METABOLISM PROGRAMP30CA043703 · NCI · CASE WESTERN RESERVE UNIVERSITY · PI Amar Desai · 1987 to 2026
$142.3MMEDICAL SCIENTIST TRAINING PROGRAMT32GM007250 · NIGMS · CASE WESTERN RESERVE UNIVERSITY · PI HUANG, ALEX YEE-CHEN · 1985 to 2023
$33.4MRobust mapping of chromatin loops from sparse or single cell Hi-C data with DeepLoopR01HG009658 · NHGRI · CASE WESTERN RESERVE UNIVERSITY · PI Fulai Jin · 2017 to 2026
$5.1MElucidating human beta cell transcriptional regulome with low-input genomic technologiesR01DK113185 · NIDDK · CASE WESTERN RESERVE UNIVERSITY · PI Yan Li · 2018 to 2026
$4.0MHuman iPSC and glial chimeric modeling of Pelizaeus-Merzbacher DiseaseR01NS093357 · NINDS · CASE WESTERN RESERVE UNIVERSITY · PI TESAR, PAUL JOSEPH, WINDREM, MARTHA SHERRILL · 2015 to 2018
$1.5MAcquisition of a Leica TCS SP5 Confocal Microscope For A Multi-User FacilityS10OD016164 · OD · CASE WESTERN RESERVE UNIVERSITY · PI CONRAD, PATRICIA A · 2013 to 2013
$597kNCI NIH HHS P30 CA043703NHGRI NIH HHS R01 HG009658NIGMS NIH HHS T32 GM007250NIH HHS S10 OD016164NINDS NIH HHS R01 NS093357
Abstract
Pelizaeus-Merzbacher disease (PMD) is a fatal X-linked disorder caused by loss of myelinating oligodendrocytes and consequent hypomyelination. The underlying cellular and molecular dysfunctions are not fully defined, but therapeutic enhancement of oligodendrocyte survival could restore functional myelination in patients. Here we generated pure, scalable quantities of induced pluripotent stem cell-derived oligodendrocyte progenitor cells (OPCs) from a severe mouse model of PMD, Plp1
Indexed as
AnimalsCells, CulturedCell SurvivalDisease Models, AnimalEndoplasmic Reticulum StressHumansInduced Pluripotent Stem CellsMiceMutationMyelin SheathOligodendrocyte Precursor CellsOligodendrogliaPelizaeus-Merzbacher DiseaseTranscriptomeendoplasmic reticulum stresshigh-throughput screeningiPSC disease modelingmyelinoligodendrocyte progenitor cellsoligodendrocytesPelizaeus-Merzbacher diseasePLP1proteolipid protein 1rare disease
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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.