ReviewDevelopmental dynamics : an official publication of the American Association of Anatomists2020
New frontiers in modeling tuberous sclerosis with human stem cell-derived neurons and brain organoids.
Review in Developmental dynamics : an official publication of the American Association of Anatomists, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 20 papers.
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Who cites it
20 citing papers in PubMed.
- Adrenomedullin restores the human cortical interneurons migration defects induced by hypoxia.eLife · 2026Article
- Article
- Tumors and their microenvironments: Learning from pediatric brain pathologies.Biochimica et biophysica acta. Reviews on cancer · 2025Review
- Review
- Non-invasive label-free imaging analysis pipeline for in situ characterization of 3D brain organoids.Scientific reports · 2024Article
- Single cell spatial biology over developmental time can decipher pediatric brain pathologies.Neurobiology of disease · 2024Review
- Non-Invasive Label-free Analysis Pipeline for In Situ Characterization of Differentiation in 3D Brain Organoid Models.Research square · 2024Article
- Translatome analysis of tuberous sclerosis complex 1 patient-derived neural progenitor cells reveals rapamycin-dependent and independent alterations.Molecular autism · 2023Article
- The generation and properties of human cortical organoids as a disease model for malformations of cortical development.Neural regeneration research · 2023Review
- Dopamine neuron morphology and output are differentially controlled by mTORC1 and mTORC2.eLife · 2022Article
- Review
- RHOA signaling defects result in impaired axon guidance in iPSC-derived neurons from patients with tuberous sclerosis complex.Nature communications · 2021Article
- Revisiting Brain Tuberous Sclerosis Complex in Rat and Human: Shared Molecular and Cellular Pathology Leads to Distinct Neurophysiological and Behavioral Phenotypes.Neurotherapeutics : the journal of the American Society for Experimental NeuroTherapeutics · 2021Review
- Current Approaches and Future Directions for the Treatment of mTORopathies.Developmental neuroscience · 2021Review
- Human Brain Organoid Models of Developmental Epilepsies.Epilepsy currents · 2020Article
- The Lysosome at the Intersection of Cellular Growth and Destruction.Developmental cell · 2020Review
- Human in vitro models for understanding mechanisms of autism spectrum disorder.Molecular autism · 2020Review
- Recent advances in human stem cell-based modeling of Tuberous Sclerosis Complex.Molecular autism · 2020Review
- High glucose concentrations mask cellular phenotypes in a stem cell model of tuberous sclerosis complex.Epilepsy & behavior : E&B · 2019Article
- iPSCs-Based Neural 3D Systems: A Multidimensional Approach for Disease Modeling and Drug Discovery.Cells · 2019Review
Corrections and comments
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2 authors.
Funding
Abstract
Recent advances in human stem cell and genome engineering have enabled the generation of genetically defined human cellular models for brain disorders. These models can be established from a patient's own cells and can be genetically engineered to generate isogenic, controlled systems for mechanistic studies. Given the challenges of obtaining and working with primary human brain tissue, these models fill a critical gap in our understanding of normal and abnormal human brain development and provide an important complement to animal models. Recently, there has been major progress in modeling the neuropathophysiology of the canonical "mTORopathy" tuberous sclerosis complex (TSC) with such approaches. Studies using two- and three-dimensional cultures of human neurons and glia have provided new insights into how mutations in the TSC1 and TSC2 genes impact human neural development and function. Here we discuss recent progress in human stem cell-based modeling of TSC and highlight challenges and opportunities for further efforts in this area.
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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.