Human induced pluripotent stem cells for modelling neurodevelopmental disorders

Transcriptome Analysis of the Human Striatum in Tourette Syndrome

(2016) Jessica B. Lennington et al.   BIOLOGICAL PSYCHIATRY

Advances in Zika Virus Research: Stem Cell Models, Challenges, and Opportunities

(2016) Guo-li Ming et al.   Cell Stem Cell

Zika Virus Depletes Neural Progenitors in Human Cerebral Organoids through Activation of the Innate Immune Receptor TLR3

(2016) Jason Dang et al.   Cell Stem Cell

Zika Virus Infects Human Cortical Neural Progenitors and Attenuates Their Growth

(2016) Hengli Tang et al.   Cell Stem Cell

Zika Virus NS4A and NS4B Proteins Deregulate Akt-mTOR Signaling in Human Fetal Neural Stem Cells to Inhibit Neurogenesis and Induce Autophagy

(2016) Qiming Liang et al.   Cell Stem Cell

The search for true numbers of neurons and glial cells in the human brain: A review of 150 years of cell counting

(2016) Christopher S. von Bartheld et al.   JOURNAL OF COMPARATIVE NEUROLOGY

Neurodevelopmental origins of bipolar disorder: iPSC models

(2016) K. Sue O'Shea et al.   MOLECULAR AND CELLULAR NEUROSCIENCE

Deriving human ENS lineages for cell therapy and drug discovery in Hirschsprung disease

(2016) Faranak Fattahi et al.   NATURE

A human neurodevelopmental model for Williams syndrome

(2016) Thanathom Chailangkarn et al.   NATURE

Autism-like behaviours and germline transmission in transgenic monkeys overexpressing MeCP2

(2016) Zhen Liu et al.   NATURE

Molecular signatures associated with ZIKV exposure in human cortical neural progenitors

(2016) Feiran Zhang et al.   NUCLEIC ACIDS RESEARCH

KCC2 rescues functional deficits in human neurons derived from patients with Rett syndrome

(2016) Xin Tang et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Zika virus impairs growth in human neurospheres and brain organoids

(2016) P. P. Garcez et al.   SCIENCE

Establishment of Reporter Lines for Detecting Fragile X Mental Retardation (FMR1) Gene Reactivation in Human Neural Cells

(2016) Meng Li et al.   STEM CELLS

Whole-genome mutational burden analysis of three pluripotency induction methods

(2016) Kunal Bhutani et al.   Nature Communications

Comparative Analysis Between Flaviviruses Reveals Specific Neural Stem Cell Tropism for Zika Virus in the Mouse Developing Neocortex

(2016) Jean-Baptiste Brault et al.   EBioMedicine

Zika Virus Strains Potentially Display Different Infectious Profiles in Human Neural Cells

(2016) Yannick Simonin et al.   EBioMedicine

FOXG1-Dependent Dysregulation of GABA/Glutamate Neuron Differentiation in Autism Spectrum Disorders

(2015) Jessica Mariani et al.   CELL

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

(2015) Jerome Mertens et al.   Cell Stem Cell

Imbalance of excitatory/inhibitory synaptic protein expression in iPSC-derived neurons from FOXG1 +/− patients and in foxg1 +/− mice

(2015) Tommaso Patriarchi et al.   EUROPEAN JOURNAL OF HUMAN GENETICS

Altered microtubule dynamics and vesicular transport in mouse and human MeCP2-deficient astrocytes

(2015) Chloé Delépine et al.   HUMAN MOLECULAR GENETICS

Altered neuronal network and rescue in a human MECP2 duplication model

(2015) S Nageshappa et al.   MOLECULAR PSYCHIATRY

Characterization of bipolar disorder patient-specific induced pluripotent stem cells from a family reveals neurodevelopmental and mRNA expression abnormalities

(2015) J M Madison et al.   MOLECULAR PSYCHIATRY

Differential responses to lithium in hyperexcitable neurons from patients with bipolar disorder

(2015) Jerome Mertens et al.   NATURE

A comparison of genetically matched cell lines reveals the equivalence of human iPSCs and ESCs

(2015) Jiho Choi et al.   NATURE BIOTECHNOLOGY

Mapping DNA methylation across development, genotype and schizophrenia in the human frontal cortex

(2015) Andrew E Jaffe et al.   NATURE NEUROSCIENCE

Current status of pluripotent stem cells: moving the first therapies to the clinic

(2015) Erin A. Kimbrel et al.   NATURE REVIEWS DRUG DISCOVERY

Altered epigenetic regulation in early development associated with schizophrenia

(2015) Hemi Malkki   Nature Reviews Neurology

Applications of CRISPR–Cas systems in neuroscience

(2015) Matthias Heidenreich et al.   NATURE REVIEWS NEUROSCIENCE

MECP2e1 isoform mutation affects the form and function of neurons derived from Rett syndrome patient iPS cells

(2015) Ugljesa Djuric et al.   NEUROBIOLOGY OF DISEASE

The autism-associated chromatin modifier CHD8 regulates other autism risk genes during human neurodevelopment

(2015) Justin Cotney et al.   Nature Communications

Reversion of FMR1 Methylation and Silencing by Editing the Triplet Repeats in Fragile X iPSC-Derived Neurons

(2015) Chul-Yong Park et al.   Cell Reports

Human induced pluripotent stem cell derived neurons as a model for Williams-Beuren syndrome

(2015) Shahryar Khattak et al.   Molecular Brain

High-Throughput Screening to Identify Compounds That Increase Fragile X Mental Retardation Protein Expression in Neural Stem Cells Differentiated From Fragile X Syndrome Patient-Derived Induced Pluripotent Stem Cells

(2015) Daman Kumari et al.   Stem Cells Translational Medicine

CRISPR/Cas9-mediated heterozygous knockout of the autism gene CHD8 and characterization of its transcriptional networks in neurodevelopment

(2015) Ping Wang et al.   Molecular Autism

Increased abundance of translation machinery in stem cell–derived neural progenitor cells from four schizophrenia patients

(2015) A Topol et al.   Translational Psychiatry

Molecular Mechanisms Regulating the Defects in Fragile X Syndrome Neurons Derived from Human Pluripotent Stem Cells

(2015) Tomer Halevy et al.   Stem Cell Reports

MicroRNA Profiling of Neurons Generated Using Induced Pluripotent Stem Cells Derived from Patients with Schizophrenia and Schizoaffective Disorder, and 22q11.2 Del

(2015) Dejian Zhao et al.   PLoS One

Cellular and Molecular Mechanisms of Synaptic Specificity

(2014) Shaul Yogev et al.   Annual Review of Cell and Developmental Biology

Disruptive CHD8 Mutations Define a Subtype of Autism Early in Development

(2014) Raphael Bernier et al.   CELL

Modeling a Genetic Risk for Schizophrenia in iPSCs and Mice Reveals Neural Stem Cell Deficits Associated with Adherens Junctions and Polarity

(2014) Ki-Jun Yoon et al.   Cell Stem Cell

GluD1 is a common altered player in neuronal differentiation from both MECP2-mutated and CDKL5-mutated iPS cells

(2014) Gabriella Livide et al.   EUROPEAN JOURNAL OF HUMAN GENETICS

Mutant astrocytes differentiated from Rett syndrome patients-specific iPSCs have adverse effects on wild-type neurons

(2014) Emily Cunningham Williams et al.   HUMAN MOLECULAR GENETICS

Modeling non-syndromic autism and the impact of TRPC6 disruption in human neurons

(2014) K Griesi-Oliveira et al.   MOLECULAR PSYCHIATRY

Phenotypic differences in hiPSC NPCs derived from patients with schizophrenia

(2014) K Brennand et al.   MOLECULAR PSYCHIATRY

Synaptic dysregulation in a human iPS cell model of mental disorders

(2014) Zhexing Wen et al.   NATURE

Biological insights from 108 schizophrenia-associated genetic loci

(2014)   NATURE

CRISPR-Cas systems for editing, regulating and targeting genomes

(2014) Jeffry D Sander et al.   NATURE BIOTECHNOLOGY

Loss of mTOR-Dependent Macroautophagy Causes Autistic-like Synaptic Pruning Deficits

(2014) Guomei Tang et al.   NEURON

A Quantitative Framework to Evaluate Modeling of Cortical Development by Neural Stem Cells

(2014) Jason L. Stein et al.   NEURON

Increased L1 Retrotransposition in the Neuronal Genome in Schizophrenia

(2014) Miki Bundo et al.   NEURON

Patches of Disorganization in the Neocortex of Children with Autism

(2014) Rich Stoner et al.   NEW ENGLAND JOURNAL OF MEDICINE

CHD8regulates neurodevelopmental pathways associated with autism spectrum disorder in neural progenitors

(2014) Aarathi Sugathan et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

iPSC-Derived Forebrain Neurons from FXS Individuals Show Defects in Initial Neurite Outgrowth

(2014) Matthew E. Doers et al.   STEM CELLS AND DEVELOPMENT

Alteration in basal and depolarization induced transcriptional network in iPSC derived neurons from Timothy syndrome

(2014) Yuan Tian et al.   Genome Medicine

Transcripts involved in calcium signaling and telencephalic neuronal fate are altered in induced pluripotent stem cells from bipolar disorder patients

(2014) H M Chen et al.   Translational Psychiatry

Human iPSC Neurons Display Activity-Dependent Neurotransmitter Secretion: Aberrant Catecholamine Levels in Schizophrenia Neurons

(2014) Vivian Hook et al.   Stem Cell Reports

Modeling Hippocampal Neurogenesis Using Human Pluripotent Stem Cells

(2014) Diana Xuan Yu et al.   Stem Cell Reports

The aging signature: a hallmark of induced pluripotent stem cells?

(2013) Leili Rohani et al.   AGING CELL

Spatial and Temporal Mapping of De Novo Mutations in Schizophrenia to a Fetal Prefrontal Cortical Network

(2013) Suleyman Gulsuner et al.   CELL

Coexpression Networks Implicate Human Midfetal Deep Cortical Projection Neurons in the Pathogenesis of Autism

(2013) A. Jeremy Willsey et al.   CELL

Integrative Functional Genomic Analyses Implicate Specific Molecular Pathways and Circuits in Autism

(2013) Neelroop N. Parikshak et al.   CELL

Epigenetics of Reprogramming to Induced Pluripotency

(2013) Bernadett Papp et al.   CELL

Human iPSC-Based Modeling of Late-Onset Disease via Progerin-Induced Aging

(2013) Justine D. Miller et al.   Cell Stem Cell

Directed Differentiation and Functional Maturation of Cortical Interneurons from Human Embryonic Stem Cells

(2013) Asif M. Maroof et al.   Cell Stem Cell

Abnormal neuronal differentiation and mitochondrial dysfunction in hair follicle-derived induced pluripotent stem cells of schizophrenia patients

(2013) O Robicsek et al.   MOLECULAR PSYCHIATRY

Cerebral organoids model human brain development and microcephaly

(2013) Madeline A. Lancaster et al.   NATURE

Genome-wide association analysis identifies 13 new risk loci for schizophrenia

(2013) Stephan Ripke et al.   NATURE GENETICS

Timothy syndrome is associated with activity-dependent dendritic retraction in rodent and human neurons

(2013) Jocelyn F Krey et al.   NATURE NEUROSCIENCE

Drug discovery for autism spectrum disorder: challenges and opportunities

(2013) Anirvan Ghosh et al.   NATURE REVIEWS DRUG DISCOVERY

Mechanisms and models of somatic cell reprogramming

(2013) Yosef Buganim et al.   NATURE REVIEWS GENETICS

Nuclear calcium signalling in the regulation of brain function

(2013) Hilmar Bading   NATURE REVIEWS NEUROSCIENCE

Self-organization of axial polarity, inside-out layer pattern, and species-specific progenitor dynamics in human ES cell-derived neocortex

(2013) T. Kadoshima et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Fragile X mental retardation protein and synaptic plasticity

(2013) Michael S Sidorov et al.   Molecular Brain

Focal cortical dysplasias in autism spectrum disorders

(2013) Manuel F Casanova et al.   Acta Neuropathologica Communications

Embryonic stem cell and induced pluripotent stem cell: an epigenetic perspective

(2012) Gaoyang Liang et al.   CELL RESEARCH

Discovery of Inhibitors of Microglial Neurotoxicity Acting Through Multiple Mechanisms Using a Stem-Cell-Based Phenotypic Assay

(2012) Susanne Höing et al.   Cell Stem Cell

Low Incidence of DNA Sequence Variation in Human Induced Pluripotent Stem Cells Generated by Nonintegrating Plasmid Expression

(2012) Linzhao Cheng et al.   Cell Stem Cell

Erosion of Dosage Compensation Impacts Human iPSC Disease Modeling

(2012) Shila Mekhoubad et al.   Cell Stem Cell

New perspectives on the biology of fragile X syndrome

(2012) Tao Wang et al.   CURRENT OPINION IN GENETICS & DEVELOPMENT

Reduction of BDNF expression inFmr1knockout mice worsens cognitive deficits but improves hyperactivity and sensorimotor deficits

(2012) M. Uutela et al.   GENES BRAIN AND BEHAVIOR

Fragile X syndrome: causes, diagnosis, mechanisms, and therapeutics

(2012) Claudia Bagni et al.   JOURNAL OF CLINICAL INVESTIGATION

Increased inflammatory markers identified in the dorsolateral prefrontal cortex of individuals with schizophrenia

(2012) S G Fillman et al.   MOLECULAR PSYCHIATRY

Somatic copy number mosaicism in human skin revealed by induced pluripotent stem cells

(2012) Alexej Abyzov et al.   NATURE

De novo mutations revealed by whole-exome sequencing are strongly associated with autism

(2012) Stephan J. Sanders et al.   NATURE

Sporadic autism exomes reveal a highly interconnected protein network of de novo mutations

(2012) Brian J. O’Roak et al.   NATURE

Large-scale screening using familial dysautonomia induced pluripotent stem cells identifies compounds that rescue IKBKAP expression

(2012) Gabsang Lee et al.   NATURE BIOTECHNOLOGY

CDKL5 ensures excitatory synapse stability by reinforcing NGL-1–PSD95 interaction in the postsynaptic compartment and is impaired in patient iPSC-derived neurons

(2012) Sara Ricciardi et al.   NATURE CELL BIOLOGY

Human cerebral cortex development from pluripotent stem cells to functional excitatory synapses

(2012) Yichen Shi et al.   NATURE NEUROSCIENCE

Structural plasticity upon learning: regulation and functions

(2012) Pico Caroni et al.   NATURE REVIEWS NEUROSCIENCE

Derivation of autism spectrum disorder-specific induced pluripotent stem cells from peripheral blood mononuclear cells

(2012) Brooke A. DeRosa et al.   NEUROSCIENCE LETTERS

The GABA Excitatory/Inhibitory Shift in Brain Maturation and Neurological Disorders

(2012) Yehezkel Ben-Ari et al.   NEUROSCIENTIST

Modeling human cortical development in vitro using induced pluripotent stem cells

(2012) J. Mariani et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Multiplex Targeted Sequencing Identifies Recurrently Mutated Genes in Autism Spectrum Disorders

(2012) B. J. O'Roak et al.   SCIENCE

Drug Screening for ALS Using Patient-Specific Induced Pluripotent Stem Cells

(2012) N. Egawa et al.   Science Translational Medicine

Prevention of β-amyloid induced toxicity in human iPS cell-derived neurons by inhibition of Cyclin-dependent kinases and associated cell cycle events

(2012) Xiaohong Xu et al.   Stem Cell Research

Synapse dysfunction in autism: a molecular medicine approach to drug discovery in neurodevelopmental disorders

(2012) Will Spooren et al.   TRENDS IN PHARMACOLOGICAL SCIENCES

Molecular analysis of FMR1 reactivation in fragile-X induced pluripotent stem cells and their neuronal derivatives

(2012) Ori Bar-Nur et al.   Journal of Molecular Cell Biology

Synaptic Dysfunction in Neurodevelopmental Disorders Associated with Autism and Intellectual Disabilities

(2012) H. Y. Zoghbi et al.   Cold Spring Harbor Perspectives in Biology

X-Chromosome Inactivation in Rett Syndrome Human Induced Pluripotent Stem Cells

(2012) Aaron Y. L. Cheung et al.   Frontiers in Psychiatry

Genetic Heritability and Shared Environmental Factors Among Twin Pairs With Autism

(2011) Joachim Hallmayer   ARCHIVES OF GENERAL PSYCHIATRY

The Human Brain in a Dish: The Promise of iPSC-Derived Neurons

(2011) Ricardo Dolmetsch et al.   CELL

Reprogramming Factor Stoichiometry Influences the Epigenetic State and Biological Properties of Induced Pluripotent Stem Cells

(2011) Bryce W. Carey et al.   Cell Stem Cell

Highly Efficient miRNA-Mediated Reprogramming of Mouse and Human Somatic Cells to Pluripotency

(2011) Frederick Anokye-Danso et al.   Cell Stem Cell

Constitutive heterochromatin reorganization during somatic cell reprogramming

(2011) Eden Fussner et al.   EMBO JOURNAL

iPS cells to model CDKL5-related disorders

(2011) Mariangela Amenduni et al.   EUROPEAN JOURNAL OF HUMAN GENETICS

Rejuvenating senescent and centenarian human cells by reprogramming through the pluripotent state

(2011) L. Lapasset et al.   GENES & DEVELOPMENT

Isolation of MECP2-null Rett Syndrome patient hiPS cells and isogenic controls through X-chromosome inactivation

(2011) Aaron Y.L. Cheung et al.   HUMAN MOLECULAR GENETICS

Spatio-temporal transcriptome of the human brain

(2011) Hyo Jung Kang et al.   NATURE

Temporal dynamics and genetic control of transcription in the human prefrontal cortex

(2011) Carlo Colantuoni et al.   NATURE

Modelling schizophrenia using human induced pluripotent stem cells

(2011) Kristen J. Brennand et al.   NATURE

Somatic coding mutations in human induced pluripotent stem cells

(2011) Athurva Gore et al.   NATURE

Transcriptomic analysis of autistic brain reveals convergent molecular pathology

(2011) Irina Voineagu et al.   NATURE

Exome sequencing supports a de novo mutational paradigm for schizophrenia

(2011) Bin Xu et al.   NATURE GENETICS

Genome-wide association study identifies five new schizophrenia loci

(2011)   NATURE GENETICS

Exome sequencing in sporadic autism spectrum disorders identifies severe de novo mutations

(2011) Brian J O'Roak et al.   NATURE GENETICS

Using iPSC-derived neurons to uncover cellular phenotypes associated with Timothy syndrome

(2011) Sergiu P Paşca et al.   NATURE MEDICINE

Constructing and Deconstructing Stem Cell Models of Neurological Disease

(2011) Steve S.W. Han et al.   NEURON

Rare De Novo and Transmitted Copy-Number Variation in Autistic Spectrum Disorders

(2011) Dan Levy et al.   NEURON

Multiple Recurrent De Novo CNVs, Including Duplications of the 7q11.23 Williams Syndrome Region, Are Strongly Associated with Autism

(2011) Stephan J. Sanders et al.   NEURON

Isogenic Pairs of Wild Type and Mutant Induced Pluripotent Stem Cell (iPSC) Lines from Rett Syndrome Patients as In Vitro Disease Model

(2011) Gene Ananiev et al.   PLoS One

Epigenetic Characterization of the FMR1 Gene and Aberrant Neurodevelopment in Human Induced Pluripotent Stem Cell Models of Fragile X Syndrome

(2011) Steven D. Sheridan et al.   PLoS One

Neuronal maturation defect in induced pluripotent stem cells from patients with Rett syndrome

(2011) K.-Y. Kim et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Concise Review: The Promise of Human Induced Pluripotent Stem Cell-Based Studies of Schizophrenia

(2011) Kristen J. Brennand et al.   STEM CELLS

Global epigenetic changes during somatic cell reprogramming to iPS cells

(2011) Anna Mattout et al.   Journal of Molecular Cell Biology

Rett syndrome: Revised diagnostic criteria and nomenclature

(2010) Jeffrey L. Neul et al.   ANNALS OF NEUROLOGY

Genetics and Cell Biology of Building Specific Synaptic Connectivity

(2010) Kang Shen et al.   Annual Review of Neuroscience

A Model for Neural Development and Treatment of Rett Syndrome Using Human Induced Pluripotent Stem Cells

(2010) Maria C.N. Marchetto et al.   CELL

Female Human iPSCs Retain an Inactive X Chromosome

(2010) Jason Tchieu et al.   Cell Stem Cell

Differential Modeling of Fragile X Syndrome by Human Embryonic Stem Cells and Induced Pluripotent Stem Cells

(2010) Achia Urbach et al.   Cell Stem Cell

Chromatin Structure and Gene Expression Programs of Human Embryonic and Induced Pluripotent Stem Cells

(2010) Matthew G. Guenther et al.   Cell Stem Cell

Molecular Analyses of Human Induced Pluripotent Stem Cells and Embryonic Stem Cells

(2010) Mark H. Chin et al.   Cell Stem Cell

L1 retrotransposition in neurons is modulated by MeCP2

(2010) Alysson R. Muotri et al.   NATURE

Functional impact of global rare copy number variation in autism spectrum disorders

(2010) Dalila Pinto et al.   NATURE

BDNF and TrkB in neuronal differentiation of Fmr1-knockout mouse

(2010) Verna Louhivuori et al.   NEUROBIOLOGY OF DISEASE

The human brain in numbers: a linearly scaled-up primate brain

(2009) Suzana Herculano-Houzel   Frontiers in Human Neuroscience

Autism genome-wide copy number variation reveals ubiquitin and neuronal genes

(2009) Joseph T. Glessner et al.   NATURE

Highly efficient neural conversion of human ES and iPS cells by dual inhibition of SMAD signaling

(2009) Stuart M Chambers et al.   NATURE BIOTECHNOLOGY

Isolation of human iPS cells using EOS lentiviral vectors to select for pluripotency

(2009) Akitsu Hotta et al.   NATURE METHODS

Experience-dependent structural synaptic plasticity in the mammalian brain

(2009) Anthony Holtmaat et al.   NATURE REVIEWS NEUROSCIENCE

Functional and Evolutionary Insights into Human Brain Development through Global Transcriptome Analysis

(2009) Matthew B. Johnson et al.   NEURON

Structural Variation of Chromosomes in Autism Spectrum Disorder

(2008) Christian R. Marshall et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Molecular Cytogenetic Analysis and Resequencing of Contactin Associated Protein-Like 2 in Autism Spectrum Disorders

(2008) Betul Bakkaloglu et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Global Transcription in Pluripotent Embryonic Stem Cells

(2008) Sol Efroni et al.   Cell Stem Cell

Self-Organized Formation of Polarized Cortical Tissues from ESCs and Its Active Manipulation by Extrinsic Signals

(2008) Mototsugu Eiraku et al.   Cell Stem Cell

Dissecting direct reprogramming through integrative genomic analysis

(2008) Tarjei S. Mikkelsen et al.   NATURE

The adult human brain in preclinical drug development

(2008) Mike Dragunow   NATURE REVIEWS DRUG DISCOVERY