Synaptic dysfunction in neurodegenerative and neurodevelopmental diseases: an overview of induced pluripotent stem-cell-based disease models DOI Creative Commons
Era Taoufik,

Georgia Kouroupi,

Ourania Zygogianni

et al.

Open Biology, Journal Year: 2018, Volume and Issue: 8(9)

Published: Sept. 1, 2018

Synaptic dysfunction in CNS disorders is the outcome of perturbations physiological synapse structure and function, can be either cause or consequence specific pathologies. Accumulating data field neuropsychiatric disorders, including autism spectrum schizophrenia bipolar disorder, point to a neurodevelopmental origin these Due relatively early onset behavioural cognitive symptoms, it generally acknowledged that mental illness initiates at level. On other hand, synaptic has been considered as an endpoint incident neurodegenerative diseases, such Alzheimer's, Parkinson's Huntington's, mainly due considerably later clinical symptoms progressive appearance deficits. This dichotomy recently challenged, particularly since discovery cell reprogramming technologies generation induced pluripotent stem cells from patient somatic cells. The creation ‘disease-in-a-dish’ models for multiple pathologies revealed unexpected commonalities molecular cellular mechanisms operating both developmental degenerative conditions, most which meet In this review we discuss prototype emphasizing overlapping features synaptopathy have suggested by studies using stem-cell-based systems. These valuable disease highlighted potential component classical diseases worth pursuing investigating further. Moving demonstration correlation understanding mechanistic causality forms basis developing novel therapeutics.

Language: Английский

Complex Oscillatory Waves Emerging from Cortical Organoids Model Early Human Brain Network Development DOI Creative Commons
Cleber A. Trujillo, Richard Gao, Priscilla D. Negraes

et al.

Cell stem cell, Journal Year: 2019, Volume and Issue: 25(4), P. 558 - 569.e7

Published: Aug. 29, 2019

Language: Английский

Citations

736

Engineering of human brain organoids with a functional vascular-like system DOI
Bilal Çakir, Yangfei Xiang, Yoshiaki Tanaka

et al.

Nature Methods, Journal Year: 2019, Volume and Issue: 16(11), P. 1169 - 1175

Published: Oct. 7, 2019

Language: Английский

Citations

724

Brain organoids: advances, applications and challenges DOI Open Access
Xuyu Qian, Hongjun Song, Guo‐li Ming

et al.

Development, Journal Year: 2019, Volume and Issue: 146(8)

Published: April 15, 2019

Brain organoids are self-assembled three-dimensional aggregates generated from pluripotent stem cells with cell types and cytoarchitectures that resemble the embryonic human brain. As such, they have emerged as novel model systems can be used to investigate brain development disorders. Although mimic many key features of early at molecular, cellular, structural functional levels, some aspects development, such formation distinct cortical neuronal layers, gyrification, establishment complex circuitry, not fully recapitulated. Here, we summarize recent advances in organoid methodologies discuss their applications disease modeling. In addition, compare current brain, highlighting currently cannot recapitulated, perspectives for advancing technologies expand applications.

Language: Английский

Citations

549

Reliability of human cortical organoid generation DOI
Se‐Jin Yoon,

Lubayna S. Elahi,

Anca M. Pașca

et al.

Nature Methods, Journal Year: 2018, Volume and Issue: 16(1), P. 75 - 78

Published: Dec. 14, 2018

Language: Английский

Citations

416

Generation of Functional Human 3D Cortico-Motor Assembloids DOI Creative Commons
Jimena Andersen, Omer Revah, Yuki Miura

et al.

Cell, Journal Year: 2020, Volume and Issue: 183(7), P. 1913 - 1929.e26

Published: Dec. 1, 2020

Language: Английский

Citations

414

hESC-Derived Thalamic Organoids Form Reciprocal Projections When Fused with Cortical Organoids DOI Creative Commons
Yangfei Xiang, Yoshiaki Tanaka,

Bilal Çakır

et al.

Cell stem cell, Journal Year: 2019, Volume and Issue: 24(3), P. 487 - 497.e7

Published: Feb. 23, 2019

Language: Английский

Citations

406

Generation and assembly of human brain region–specific three-dimensional cultures DOI
Steven A. Sloan, Jimena Andersen, Anca M. Pașca

et al.

Nature Protocols, Journal Year: 2018, Volume and Issue: 13(9), P. 2062 - 2085

Published: Sept. 1, 2018

Language: Английский

Citations

369

Human CNS barrier-forming organoids with cerebrospinal fluid production DOI
Laura Pellegrini, Claudia Bonfio, Jessica Chadwick

et al.

Science, Journal Year: 2020, Volume and Issue: 369(6500)

Published: June 11, 2020

Cerebrospinal fluid (CSF) is a vital liquid, providing nutrients and signaling molecules clearing out toxic by-products from the brain. The CSF produced by choroid plexus (ChP), protective epithelial barrier that also prevents free entry of or drugs blood. Here, we establish human ChP organoids with selective CSF-like secretion in self-contained compartments. We show this vitro exhibits same selectivity to small as vivo ChP-CSF can predict central nervous system (CNS) permeability new compounds. transcriptomic proteomic signatures reveal high degree similarity vivo. Finally, intersection single-cell transcriptomics analysis uncovers key components previously unidentified specialized subtypes.

Language: Английский

Citations

357

Generation of human striatal organoids and cortico-striatal assembloids from human pluripotent stem cells DOI
Yuki Miura, Min-Yin Li, Fikri Birey

et al.

Nature Biotechnology, Journal Year: 2020, Volume and Issue: 38(12), P. 1421 - 1430

Published: Dec. 1, 2020

Language: Английский

Citations

323

Differentiation and maturation of oligodendrocytes in human three-dimensional neural cultures DOI
Rebecca M. Marton, Yuki Miura, Steven A. Sloan

et al.

Nature Neuroscience, Journal Year: 2019, Volume and Issue: 22(3), P. 484 - 491

Published: Jan. 28, 2019

Language: Английский

Citations

306