Novel human iPSC models of neuroinflammation in neurodegenerative disease and regenerative medicine

Induction of Pluripotent Stem Cells from Adult Human Fibroblasts by Defined Factors

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Neurotoxic reactive astrocytes are induced by activated microglia

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Cerebral organoids model human brain development and microcephaly

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New insights into the genetic etiology of Alzheimer’s disease and related dementias

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Multiple sclerosis

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Brain-Region-Specific Organoids Using Mini-bioreactors for Modeling ZIKV Exposure

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Microglia states and nomenclature: A field at its crossroads

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Perturb-Seq: Dissecting Molecular Circuits with Scalable Single-Cell RNA Profiling of Pooled Genetic Screens

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Microglia in neurodegeneration

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Role of neuroinflammation in neurodegeneration development

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Microglia in neurodegenerative diseases: mechanism and potential therapeutic targets

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Multiple sclerosis genomic map implicates peripheral immune cells and microglia in susceptibility

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Multiplexed droplet single-cell RNA-sequencing using natural genetic variation

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iPSC-derived human microglia-like cells to study neurological diseases

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Engineering of human brain organoids with a functional vascular-like system

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Dual microglia effects on blood brain barrier permeability induced by systemic inflammation

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Derivation of blood-brain barrier endothelial cells from human pluripotent stem cells

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A lymphocyte–microglia–astrocyte axis in chronic active multiple sclerosis

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Directly Reprogrammed Human Neurons Retain Aging-Associated Transcriptomic Signatures and Reveal Age-Related Nucleocytoplasmic Defects.

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A Gradient of neuronal loss and meningeal inflammation in multiple sclerosis

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The blood-brain barrier in aging and neurodegeneration

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The Incidence of Parkinson's Disease: A Systematic Review and Meta-Analysis

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Efficient derivation of microglia-like cells from human pluripotent stem cells

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Human iPSC-Derived Blood-Brain Barrier Chips Enable Disease Modeling and Personalized Medicine Applications

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Microglia innately develop within cerebral organoids

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A highly efficient human pluripotent stem cell microglia model displays a neuronal-co-culture-specific expression profile and inflammatory response

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Human iPSC-Derived Oligodendrocyte Progenitor Cells Can Myelinate and Rescue a Mouse Model of Congenital Hypomyelination

Wang, S., Bates, J., Li, X., Schanz, S., Chandler-Militello, D., Levine, C., Maherali, N., Studer, L., Hochedlinger, K., Windrem, M., et al. (2013) · Cell Stem Cell · doi:10.1016/j.stem.2012.12.002 · Cited by 586 · see citing works

Neuroinflammation in the central nervous system: Symphony of glial cells

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Maturation and circuit integration of transplanted human cortical organoids

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APOE4 impairs myelination via cholesterol dysregulation in oligodendrocytes

Blanchard, J. W., Akay, L. A., Davila-Velderrain, J., von Maydell, D., Mathys, H., Davidson, S. M., Effenberger, A., Chen, C.-Y., Maner-Smith, K., Hajjar, I., et al. (2022) · Nature · doi:10.1038/s41586-022-05439-w · Cited by 493 · see citing works

Induced pluripotent stem cells (iPSCs): molecular mechanisms of induction and applications

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Derivation and Expansion Using Only Small Molecules of Human Neural Progenitors for Neurodegenerative Disease Modeling

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An Efficient Platform for Astrocyte Differentiation from Human Induced Pluripotent Stem Cells

TCW, J., Wang, M., Pimenova, A. A., Bowles, K. R., Hartley, B. J., Lacin, E., Machlovi, S. I., Abdelaal, R., Karch, C. M., Phatnani, H., et al. (2017) · Stem Cell Reports · doi:10.1016/j.stemcr.2017.06.018 · Cited by 435 · see citing works

Astrocyte contribution to dysfunction, risk and progression in neurodegenerative disorders

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Generation of vascularized brain organoids to study neurovascular interactions

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α-Synuclein-specific T cell reactivity is associated with preclinical and early Parkinson’s disease

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Development of a Chimeric Model to Study and Manipulate Human Microglia In Vivo

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Patient-Specific iPSC-Derived Astrocytes Contribute to Non-Cell-Autonomous Neurodegeneration in Parkinson's Disease

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Microglia ferroptosis is regulated by SEC24B and contributes to neurodegeneration

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Differentiation and maturation of oligodendrocytes in human three-dimensional neural cultures

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Induction of myelinating oligodendrocytes in human cortical spheroids

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Meningeal Immunity and Its Function in Maintenance of the Central Nervous System in Health and Disease

Alves de Lima, K., Rustenhoven, J., & Kipnis, J. (2020) · Annual Review of Immunology · doi:10.1146/annurev-immunol-102319-103410 · Cited by 348 · see citing works

Blood-brain-barrier spheroids as an in vitro screening platform for brain-penetrating agents

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A nomenclature consensus for nervous system organoids and assembloids

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An in vivo neuroimmune organoid model to study human microglia phenotypes

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Human cerebral organoids — a new tool for clinical neurology research

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Differentiation of human oligodendrocytes from pluripotent stem cells

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A CRISPRi/a platform in human iPSC-derived microglia uncovers regulators of disease states

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Rapid and efficient generation of oligodendrocytes from human induced pluripotent stem cells using transcription factors

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Locus for severity implicates CNS resilience in progression of multiple sclerosis

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Fully defined human pluripotent stem cell-derived microglia and tri-culture system model C3 production in Alzheimer’s disease

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Generation and isolation of oligodendrocyte progenitor cells from human pluripotent stem cells

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iPS-cell-derived microglia promote brain organoid maturation via cholesterol transfer

Park, D. S., Kozaki, T., Tiwari, S. K., Moreira, M., Khalilnezhad, A., Torta, F. T., Olivié, N., Thiam, C. H., Liani, O., Silvin, A., et al. (2023) · Nature · doi:10.1038/s41586-023-06713-1 · Cited by 237 · see citing works

NFIA is a gliogenic switch enabling rapid derivation of functional human astrocytes from pluripotent stem cells

Tchieu, J., Calder, E. L., Guttikonda, S. R., Gutzwiller, E. M., Aromolaran, K. A., Steinbeck, J. A., Goldstein, P. A., & Studer, L. (2019) · Nature Biotechnology · doi:10.1038/s41587-019-0035-0 · Cited by 231 · see citing works

Cellular senescence in progenitor cells contributes to diminished remyelination potential in progressive multiple sclerosis

Nicaise, A. M., Wagstaff, L. J., Willis, C. M., Paisie, C. A., Chandok, H., Robson, P., Fossati, V., Williams, A., & Crocker, S. J. (2019) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.1818348116 · Cited by 227 · see citing works

Crosstalk between astrocytes and microglia results in increased degradation of α-synuclein and amyloid-β aggregates

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Developing human pluripotent stem cell-based cerebral organoids with a controllable microglia ratio for modeling brain development and pathology

Xu, R., Boreland, A. J., Li, X., Erickson, C., Jin, M., Atkins, C., Pang, Z. P., Daniels, B. P., & Jiang, P. (2021) · Stem Cell Reports · doi:10.1016/j.stemcr.2021.06.011 · Cited by 222 · see citing works

Zika virus replicates in adult human brain tissue and impairs synapses and memory in mice

Figueiredo, C. P., Barros‐Aragão, F. G. Q., Neris, R. L. S., Frost, P. S., Soares, C., Souza, I. N. D. O., Zeidler, J. D., Zamberlan, D. C., de Sousa, V. L., de Souza, A. S., et al. (2019) · Nature Communications · doi:10.1038/s41467-019-11866-7 · Cited by 211 · see citing works

Human iPSC-derived microglia assume a primary microglia-like state after transplantation into the neonatal mouse brain

Svoboda, D. S., Barrasa, M. I., Shu, J., Rietjens, R. G. J., Zhang, S., Mitalipova, M., Berube, P., Fu, D., Shultz, L. D., Bell, G. W., et al. (2019) · Proceedings of the National Academy of Sciences · doi:10.1073/pnas.1913541116 · Cited by 203 · see citing works

Human iPSC-derived mature microglia retain their identity and functionally integrate in the chimeric mouse brain

Xu, R., Li, X., Boreland, A. J., Posyton, A., Kwan, K. Y., Hart, R. P., & Jiang, P. (2020) · Nature Communications · doi:10.1038/s41467-020-15411-9 · Cited by 190 · see citing works

Functionalization of Brain Region-specific Spheroids with Isogenic Microglia-like Cells

Song, L., Yuan, X., Jones, Z., Vied, C., Yu, M., Marzano, M., Hua, T., Sang, Q. A., Guan, J., Ma, T., et al. (2019) · Scientific Reports · doi:10.1038/s41598-019-47444-6 · Cited by 186 · see citing works

Metabolic alterations in Parkinson’s disease astrocytes

Sonninen, T.-M., Hämäläinen, R. H., Koskuvi, M., Oksanen, M., Shakirzyanova, A., Wojciechowski, S., Puttonen, K. A., Naumenko, N. V., Goldsteins, G., Laham-Karam, N., et al. (2020) · Scientific Reports · doi:10.1038/s41598-020-71329-8 · Cited by 181 · see citing works

Exposure of iPSC-derived human microglia to brain substrates enables the generation and manipulation of diverse transcriptional states in vitro

Dolan, M.-J., Therrien, M., Jereb, S., Kamath, T., Gazestani, V., Atkeson, T., Marsh, S. E., Goeva, A., Lojek, N. M., Murphy, S., et al. (2023) · Nature Immunology · doi:10.1038/s41590-023-01558-2 · Cited by 174 · see citing works

A multi-pronged human microglia response to Alzheimer’s disease Aβ pathology

Mancuso, R., Fattorelli, N., Martinez-Muriana, A., Davis, E., Wolfs, L., Van Den Daele, J., Geric, I., Preman, P., Serneels, L., Poovathingal, S., et al. (2022) · doi:10.1101/2022.07.07.499139 · Cited by 169 · see citing works

Stem Cell-Based Human Blood–Brain Barrier Models for Drug Discovery and Delivery

Aday, S., Cecchelli, R., Hallier-Vanuxeem, D., Dehouck, M. P., & Ferreira, L. (2016) · Trends in Biotechnology · doi:10.1016/j.tibtech.2016.01.001 · Cited by 169 · see citing works

Modeling Blood-Brain Barrier Formation and Cerebral Cavernous Malformations in human PSCs-derived organoids

Dao, L., You, Z., Lu, L., Xu, T.-Y., Sarkar, A. K., Zhu, H., Liu, M., Calandrelli, R., Yoshida, G., Lin, P., et al. (2024) · Cell Stem Cell · doi:10.1016/j.stem.2024.04.019 · Cited by 164 · see citing works

Transplantation of human neural progenitor cells secreting GDNF into the spinal cord of patients with ALS: a phase 1/2a trial

Baloh, R. H., Johnson, J. P., Avalos, P., Allred, P., Svendsen, S. P., Gowing, G., Roxas, K., Wu, A., Donahue, B., Osborne, S., et al. (2022) · Nature Medicine · doi:10.1038/s41591-022-01956-3 · Cited by 157 · see citing works

Stem cell-derived neurons reflect features of protein networks, neuropathology, and cognitive outcome of their aged human donors

Lagomarsino, V. N., Pearse, R. V., Liu, L., Hsieh, Y.-C., Fernandez, M. A., Vinton, E. A., Paull, D., Felsky, D., Tasaki, S., Gaiteri, C., et al. (2021) · Neuron · doi:10.1016/j.neuron.2021.08.003 · Cited by 151 · see citing works

Loss of function of the mitochondrial peptidase PITRM1 induces proteotoxic stress and Alzheimer’s disease-like pathology in human cerebral organoids

Maria Jose Perez, J., Ivanyuk, D., Panagiotakopoulou, V., Napoli, G. D., Kalb, S., Brunetti, D., Al‐Shaana, R., Kaeser, S. A., Fraschka, S. A. K., Jucker, M., et al. (2020) · Molecular Psychiatry · doi:10.1038/s41380-020-0807-4 · Cited by 146 · see citing works

Structural and functional integration of human forebrain organoids with the injured adult rat visual system

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Pluripotent Stem Cell-Derived Cerebral Organoids Reveal Human Oligodendrogenesis with Dorsal and Ventral Origins

Kim, H., Xu, R., Padmashri, R., Dunaevsky, A., Liu, Y., Dreyfus, C. F., & Jiang, P. (2019) · Stem Cell Reports · doi:10.1016/j.stemcr.2019.04.011 · Cited by 135 · see citing works

Intrinsic blood–brain barrier dysfunction contributes to multiple sclerosis pathogenesis

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Neural stem cell transplantation in patients with progressive multiple sclerosis: an open-label, phase 1 study

Genchi, A., Brambilla, E., Sangalli, F., Radaelli, M., Bacigaluppi, M., Furlan, R., Andolfo, A., Drago, D., Magagnotti, C., Scotti, G. M., et al. (2023) · Nature Medicine · doi:10.1038/s41591-022-02097-3 · Cited by 121 · see citing works

Human cerebral organoids establish subcortical projections in the mouse brain after transplantation

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Brain Chimeroids reveal individual susceptibility to neurotoxic triggers

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Axonal Extensions along Corticospinal Tracts from Transplanted Human Cerebral Organoids

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Correction: Derivation and Expansion Using Only Small Molecules of Human Neural Progenitors for Neurodegenerative Disease Modeling

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Microglia protect against age-associated brain pathologies

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Defects in lysosomal function and lipid metabolism in human microglia harboring a TREM2 loss of function mutation

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Comparison of the molecular profiles of human embryonic and induced pluripotent stem cells of isogenic origin

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Astrocyte-derived TNF and glutamate critically modulate microglia activation by methamphetamine

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Enhanced astrocyte responses are driven by a genetic risk allele associated with multiple sclerosis

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Profiling of microglia nodules in multiple sclerosis reveals propensity for lesion formation

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Human iPSC-derived astrocytes transplanted into the mouse brain undergo morphological changes in response to amyloid-β plaques

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“Ependymal‐in” Gradient of Thalamic Damage in Progressive Multiple Sclerosis

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Microglial inhibition alleviates alpha-synuclein propagation and neurodegeneration in Parkinson’s disease mouse model

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The complement system in neurodegenerative and inflammatory diseases of the central nervous system

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Phase I clinical trial of intracerebroventricular transplantation of allogeneic neural stem cells in people with progressive multiple sclerosis

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iPSC ‐derived microglia carrying the TREM2 R47H /+ mutation are proinflammatory and promote synapse loss

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Quantitative evaluation of brain iron accumulation in different stages of Parkinson's disease

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Generation of Human iPSC-Derived Astrocytes with a mature star-shaped phenotype for CNS modeling

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iPSC-Derived Microglia as a Model to Study Inflammation in Idiopathic Parkinson’s Disease

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Patient iPSC models reveal glia-intrinsic phenotypes in multiple sclerosis

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Increased Cholesterol Synthesis Drives Neurotoxicity in Patient Stem Cell-Derived Model of Multiple SclerosisPreprint

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A glia-enriched stem cell 3D model of the human brain mimics the glial-immune neurodegenerative phenotypes of multiple sclerosis

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Increased cholesterol synthesis drives neurotoxicity in patient stem cell-derived model of multiple sclerosis

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iPSC-derived reactive astrocytes from patients with multiple sclerosis protect cocultured neurons in inflammatory conditions

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iPSCs from people with MS can differentiate into oligodendrocytes in a homeostatic but not an inflammatory milieu

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Human Glial Chimeric Mice to Define the Role of Glial Pathology in Human Disease

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Assessing Gq-GPCR–induced human astrocyte reactivity using bioengineered neural organoids

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The role of FUT8 ‐catalyzed core fucosylation in Alzheimer's amyloid‐β oligomer‐induced activation of human microglia

Jin, L., Lucente, J. D., Mendiola, U. R., Tang, X., Zivkovic, A. M., Lebrilla, C. B., & Maezawa, I. (2023) · Glia · doi:10.1002/glia.24345 · Cited by 24 · see citing works

A Microfluidic Human Model of Blood–Brain Barrier Employing Primary Human Astrocytes

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Imaging of brain barrier inflammation and brain fluid drainage in human neurological diseases

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Single-cell sequencing reveals an important role of SPP1 and microglial activation in age-related macular degeneration

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Novel model of cortical–meningeal organoid co-culture system improves human cortical brain organoid cytoarchitecture

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MS ‐Driven Metabolic Alterations Are Recapitulated in iPSC ‐Derived Astrocytes

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Cross-species Transcriptomic Comparison of In Vitro and In Vivo Mammalian Neural Cells

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Patient iPSC models reveal glia-intrinsic phenotypes in multiple sclerosis

Clayton, B. L. L., Barbar, L., Sapar, M. L., Rusielewicz, T., Kalpana, K., Migliori, B., Paull, D., Brenner, K., Moroziewicz, D., Sand, I. K., et al. (2023) · bioRxiv · doi:10.1101/2023.08.01.551553 · Cited by 17

Comparison of two protocols for the generation of iPSC-derived human astrocytes

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Increased Cholesterol Synthesis Drivesneurotoxicity in Patient Stem Cell-Derived Model of Multiple SclerosisPreprint

Ionescu, R.-B., Nicaise, A. M., Reisz, J. A., Williams, E. C., Prasad, P., Dzieciątkowska, M., Stephenson, D., Cubero, M. S., Pirvan, L., Willis, C. M., et al. (2024) · SSRN Electronic Journal · doi:10.2139/ssrn.4735955 · Cited by 2 · see citing works

Leptomeningeal Neural Organoid (LMNO) Fusions as Models to Study Meninges-Brain SignalingPreprint

Gama, V., Jones, H., Robertson, G. L., O’Rourke, R., Romero-Morales, A. I., & Siegenthaler, J. A. (2023) · Research Square · doi:10.21203/rs.3.rs-3694849/v1 · Cited by 1 · see citing works

Leptomeningeal Neural Organoid (LMNO) Fusions as Models to Study Meninges-Brain Signaling

Jones, H. E., Robertson, G. L., Romero-Morales, A., O’Rourke, R., Siegenthaler, J. A., & Gama, V. (2023) · doi:10.1101/2023.12.01.569648

Modeling neuroinflammatory interactions between microglia and astrocytes in a human iPSC-based coculture platform

Tujula, I., Hyvärinen, T., Lotila, J., Rogal, J., Voulgaris, D., Sukki, L., Tornberg, K., Korpela, K., Jäntti, H., Malm, T., et al. (2025) · Cell Communication and Signaling · doi:10.1186/s12964-025-02304-x · Cited by 22 · see citing works

Spatially-restricted inflammation-induced senescent-like glia in multiple sclerosis and patient-derived organoids

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Smart micro/nanorobots for drug delivery in the brain

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Beyond diabetes and obesity: GLP ‐1 receptor agonists in disrupting the vicious cycle of metabolic dysfunction and neuroinflammation

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Artificial intelligence in drug discovery — what it is, where we stand and the path forward

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Age reprogramming: Innovations and ethical considerations for prolonged longevity (Review)

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Microglia from patients with multiple sclerosis display a cell-autonomous immune activation statePreprint

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Modeling neurodegenerative diseases with brain organoids: from development to disease applications

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Revisiting the Pathogenesis of X-Linked Adrenoleukodystrophy

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Elevated SGK1 increases Tau phosphorylation and microtubule instability in Alzheimer’s patient-derived cortical neurons

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Advancements, Strategies, and Challenges in Organoid-Based Drug Evaluation for Tissue Engineering and Regenerative Medicine

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The Concept of Neuroglia ‐ the State of the Art Circa 1900

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Advanced understanding of the neurotoxicity of brominated flame retardants: Integrating human neural organoids with multi-omics

Lan, Y., Gao, X., Li, W., Wang, B., Xu, L., & Li, M. (2025) · Journal of Hazardous Materials · doi:10.1016/j.jhazmat.2025.140529 · Cited by 6 · see citing works

Microglia from patients with multiple sclerosis display a cell-autonomous immune activation state

Hyvärinen, T., Lotila, J., Giudice, L., Tujula, I., Nylund, M., Ohtonen, S., Scoyni, F., Jäntti, H., Virtanen, L., Pihlava, S., et al. (2025) · Journal of Neuroinflammation · doi:10.1186/s12974-025-03575-4 · Cited by 19 · see citing works

Organoids: technology refining, current applications and future directions

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Neurological Complications in Inborn Errors of Immunity: A Scoping Review of Clinical Spectrum, Pathophysiological Mechanisms, and Therapeutic Strategies

Xiao, N., Huang, X., Chen, L., Zang, W., Guan, M., Li, T., Тузанкина, И. А., Черешнев, В. А., & Liu, G. (2025) · Clinical Reviews in Allergy & Immunology · doi:10.1007/s12016-025-09078-7 · Cited by 6 · see citing works

Deep Learning-Decoded Raman Spectroscopy for Hour-Scale iPSC Pluripotency Assessment via Lipid–Protein Biomarkers

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Brain capillary endothelial-like cells show altered barrier functionality and reduced transport of amyloid β in late-onset Alzheimer disease

Hartmann, C., Haferkamp, U., Appelt‐Menzel, A., Barenberg, J., Brachner, A., Ehrhard, T., Feldhaus, J. S., Gerhartl, A., Hollemann, T., Kulka, L. A. M., et al. (2026) · Fluids and Barriers of the CNS · doi:10.1186/s12987-025-00753-7 · Cited by 3 · see citing works

From insights to innovations: evaluating preclinical paradigms in demyelinating disease therapeutics

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Discovery of Limonoids from Xylocarpus granatum as Potential Leads against Neuroinflammation

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Tetramethylpyrazine nitrone: a multifaceted neuroprotective agent in neurodegenerative disorders

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From consortia discovery to biotech creation: An innovative approach to next‐gen Alzheimer's drugs

Giardina, S. F., Culver, A. E., Ahmed, C., Mirescu, C., Oblak, A. L., Brosch, J. R., Clayton, B., Dage, J. L., Lamb, B. T., Richardson, T. I., et al. (2026) · Alzheimer s & Dementia · doi:10.1002/alz.71165 · Cited by 2 · see citing works

Precision pharmacology of CNS GPCRs: From biased signaling to translational therapeutics

Gujjula, T. R., Bhavya, E., Gupta, R., Saini, P., Sabitha, T. B., Krishnamoorthy, P., Singh, R., Mehta, V., & Zargar, A. A. (2026) · Progress in Neuro-Psychopharmacology and Biological Psychiatry · doi:10.1016/j.pnpbp.2026.111752 · Cited by 2 · see citing works

The Dual Role of Glial Extracellular Vesicles in Neurodegeneration: Insights from iPSC-Based Models

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Neuroinflammation in Alzheimer’s and Parkinson’s diseases: pathogenic mechanisms and therapeutic strategies

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Glial-mediated pathogenesis and intervention strategies in neuromyelitis optica spectrum disorder

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Role of the WNT signalling pathway in physiological and pathological blood-brain barrier

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Targeting Active Microglia Alleviates Distal Edge of Proton Radiation-induced Neural Damage

Liao, K., Ou, D., Chen, M., Xu, F., Zhao, J., Zhou, L., Wu, R., Lin, Y., Zhang, Y., Cao, L., et al. (2025) · Advances in Radiation Oncology · doi:10.1016/j.adro.2025.101764

A human neuron-microglia tri-culture platform to study the influence of microglia on developing neuronal networks in vitroPreprint

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Adult-onset Sandhoff disease presenting with a motor neuron disease phenotype: clinical and mechanistic insights from patient-derived models

Tang, Y., Shan, D., Wang, H., Ji, X., Jiao, Y., Li, J., Zhan, Z., Liu, S., Sun, X., Lin, P., et al. (2026) · Acta Neuropathologica Communications · doi:10.1186/s40478-026-02308-9

Investigation of Neurotransplantation and Regenerative Therapy in Preclinical Models

Sugaya, K. (2026) · Contemporary clinical neuroscience · doi:10.1007/978-3-032-18296-8_28

A GPCR atlas across human microglial states in Alzheimer's disease: Insights from snRNA-seq and hiPSC models

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Generation of the RCPCMi014-A and RCPCMi014-B lines from T-lymphocytes of a healthy donor and verification of their monoclonal origin by TCR V(D)J rearrangement analysis

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