ReviewStem cell reviews and reports2014
Modeling neurodevelopmental disorders using human pluripotent stem cells.
Review in Stem cell reviews and reports, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 21 papers.
What it found
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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.
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.
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Who cites it
21 citing papers in PubMed, 43 citations in OpenAlex.
- Transcriptomic characterization of maturing neurons from human neural stem cells across developmental time points.IBRO neuroscience reports · 2025Article
- Current Applications of Human Pluripotent Stem Cells in Neuroscience Research and Cell Transplantation Therapy for Neurological Disorders.Stem cell reviews and reports · 2025Review
- Emerging roles for E3 ubiquitin ligases in neural development and disease.Frontiers in cell and developmental biology · 2025Review
- Master regulators of neurogenesis: the dynamic roles of Ephrin receptors across diverse cellular niches.Translational psychiatry · 2024Review
- Therapeutic validation and targeting of signalling networks that are dysregulated in intellectual disability.The FEBS journal · 2023Article
- Transcriptional signatures in iPSC-derived neurons are reproducible across labs when differentiation protocols are closely matched.Stem cell research · 2021Article
- Dyslexia Candidate Gene and Ciliary Gene Expression Dynamics During Human Neuronal Differentiation.Molecular neurobiology · 2020Article
- Cortical neurons derived from human pluripotent stem cells lacking FMRP display altered spontaneous firing patterns.Molecular autism · 2020Article
- Advances in Pluripotent Stem Cells: History, Mechanisms, Technologies, and Applications.Stem cell reviews and reports · 2020Review
- MKRN3 Interacts With Several Proteins Implicated in Puberty Timing but Does Not InfluenceFrontiers in endocrinology · 2019Article
- Molecular Mechanisms of Synaptic Dysregulation in Fragile X Syndrome and Autism Spectrum Disorders.Frontiers in molecular neuroscience · 2019Review
- Directed differentiation of mouse P19 embryonal carcinoma cells to neural cells in a serum- and retinoic acid-free culture medium.In vitro cellular & developmental biology. Animal · 2018Article
- RNF12 X-Linked Intellectual Disability Mutations Disrupt E3 Ligase Activity and Neural Differentiation.Cell reports · 2018Article
- Modeling Alzheimer's disease with human induced pluripotent stem (iPS) cells.Molecular and cellular neurosciences · 2016Review
- Transcriptomics analysis of iPSC-derived neurons and modeling of neuropsychiatric disorders.Molecular and cellular neurosciences · 2016Review
- Functional Deficiencies in Fragile X Neurons Derived from Human Embryonic Stem Cells.The Journal of neuroscience : the official journal of the Society for Neuroscience · 2015Article
- Molecular mechanisms regulating impaired neurogenesis of fragile X syndrome human embryonic stem cells.Stem cells and development · 2015Article
- Importance of being Nernst: Synaptic activity and functional relevance in stem cell-derived neurons.World journal of stem cells · 2015Review
- In vitro modeling of hyperpigmentation associated to neurofibromatosis type 1 using melanocytes derived from human embryonic stem cells.Proceedings of the National Academy of Sciences of the United States of America · 2015Article
- Neural stem cell replacement: a possible therapy for neurodevelopmental disorders?Neural regeneration research · 2015Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
2 authors at 1 institution in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
Neurodevelopmental disorders (NDs) are impairments that affect the development and growth of the brain and the central nervous system during embryonic and early postnatal life. Genetically manipulated animals have contributed greatly to the advancement of ND research, but many of them differ considerably from the human phenotype. Cellular in vitro models are also valuable, but the availability of human neuronal cells is limited and their lifespan in culture is short. Human pluripotent stem cells (hPSCs), including embryonic stem cells and induced pluripotent stem cells, comprise a powerful tool for studying developmentally regulated diseases, including NDs. We reviewed all recent studies in which hPSCs were used as in vitro models for diseases and syndromes characterized by impairment of neurogenesis or synaptogenesis leading to intellectual disability and delayed neurodevelopment. We analyzed their methodology and results, focusing on the data obtained following in vitro neural differentiation and gene expression and profiling of the derived neurons. Electrophysiological recording of action potentials, synaptic currents and response to neurotransmitters is pivotal for validation of the neuronal fate as well as for assessing phenotypic dysfunctions linked to the disease in question. We therefore focused on the studies which included electrophysiological recordings on the in vitro-derived neurons. Finally, we addressed specific issues that are critical for the advancement of this area of research, specifically in providing a reliable human pre-clinical research model and drug screening platform.
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Registered trials
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.