Experimental Models to Study Autism Spectrum Disorders: hiPSCs, Rodents and Zebrafish

Generation of a Novel Rat Model of Angelman Syndrome with a Complete Ube3a Gene Deletion

(2020) Andie Dodge et al.   Autism Research

Hyperactivity, Memory Defects, and Craniofacial Abnormalities in Zebrafish fmr1 Mutant Larvae

(2020) Jia Hu et al.   BEHAVIOR GENETICS

Gene replacement ameliorates deficits in mouse and human models of cyclin-dependent kinase-like 5 disorder

(2020) Yunan Gao et al.   BRAIN

De Novo ARID1B mutations cause growth delay associated with aberrant Wnt/β–catenin signaling

(2020) Xiaomin Liu et al.   HUMAN MUTATION

Oxytocin receptor signalling modulates novelty recognition but not social preference in zebrafish

(2020) Diogo Ribeiro et al.   JOURNAL OF NEUROENDOCRINOLOGY

Zebrafish as a model for studying ovarian development: Recent advances from targeted gene knockout studies

(2020) Jianzhen Li et al.   MOLECULAR AND CELLULAR ENDOCRINOLOGY

Joining forces: the need to combine science and ethics to address problems of validity and translation in neuropsychiatry research using animal models

(2020) Franck L. B. Meijboom et al.   Philosophy Ethics and Humanities in Medicine

Zebrafish as a tool to study schizophrenia-associated copy number variants

(2020) Philip D. Campbell et al.   Disease Models & Mechanisms

Impaired expression of the COSMOC/MOCOS gene unit in ASD patient stem cells

(2020) Pauline Rontani et al.   MOLECULAR PSYCHIATRY

Modeling neuronal consequences of autism-associated gene regulatory variants with human induced pluripotent stem cells

(2020) P. Joel Ross et al.   Molecular Autism

Altered spinogenesis in iPSC-derived cortical neurons from patients with autism carrying de novo SHANK3 mutations

(2019) Laura Gouder et al.   Scientific Reports

The autism/neuroprotection-linked ADNP/NAP regulate the excitatory glutamatergic synapse

(2019) Shlomo Sragovich et al.   Translational Psychiatry

Family-based exome sequencing and case-control analysis implicate CEP41 as an ASD gene

(2019) Ashok Patowary et al.   Translational Psychiatry

Scn2a haploinsufficient mice display a spectrum of phenotypes affecting anxiety, sociability, memory flexibility and ampakine CX516 rescues their hyperactivity

(2019) Tetsuya Tatsukawa et al.   Molecular Autism

Haploinsufficiency of autism causative gene Tbr1 impairs olfactory discrimination and neuronal activation of the olfactory system in mice

(2019) Tzyy-Nan Huang et al.   Molecular Autism

Intestinal dysmotility in a zebrafish (Danio rerio) shank3a;shank3b mutant model of autism

(2019) David M. James et al.   Molecular Autism

Uncovering the Functional Link Between SHANK3 Deletions and Deficiency in Neurodevelopment Using iPSC-Derived Human Neurons

(2019) Guanqun Huang et al.   Frontiers in Neuroanatomy

SHANK2 mutations associated with autism spectrum disorder cause hyperconnectivity of human neurons

(2019) Kirill Zaslavsky et al.   NATURE NEUROSCIENCE

Phenotypic Landscape of Schizophrenia-Associated Genes Defines Candidates and Their Shared Functions

(2019) Summer B. Thyme et al.   CELL

Single-cell genomics identifies cell type–specific molecular changes in autism

(2019) Dmitry Velmeshev et al.   SCIENCE

Sustained correction of associative learning deficits after brief, early treatment in a rat model of Fragile X Syndrome

(2019) Antonis Asiminas et al.   Science Translational Medicine

CNTN5-/+or EHMT2-/+human iPSC-derived neurons from individuals with autism develop hyperactive neuronal networks

(2019) Eric Deneault et al.   eLife

Synaptic dysfunction in human neurons with autism-associated deletions in PTCHD1-AS

(2019) P. Joel Ross et al.   BIOLOGICAL PSYCHIATRY

Direct reprogramming into interneurons: potential for brain repair

(2019) Maria Pereira et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Single cell analysis of autism patient with bi-allelic NRXN1-alpha deletion reveals skewed fate choice in neural progenitors and impaired neuronal functionality

(2019) Matti Lam et al.   EXPERIMENTAL CELL RESEARCH

Atypical behaviour and connectivity in SHANK3-mutant macaques

(2019) Yang Zhou et al.   NATURE

Cyfip1 haploinsufficient rats show white matter changes, myelin thinning, abnormal oligodendrocytes and behavioural inflexibility

(2019) Ana I. Silva et al.   Nature Communications

Inherited and De Novo Genetic Risk for Autism Impacts Shared Networks

(2019) Elizabeth K. Ruzzo et al.   CELL

Reelin Signaling Controls the Preference for Social Novelty in Zebrafish

(2019) Elisa Dalla Vecchia et al.   Frontiers in Behavioral Neuroscience

Use of Zebrafish in Drug Discovery Toxicology

(2019) Steven Cassar et al.   CHEMICAL RESEARCH IN TOXICOLOGY

Biallelic Mutations in TSC2 Lead to Abnormalities Associated with Cortical Tubers in Human iPSC-Derived Neurons

(2019) Kellen D. Winden et al.   JOURNAL OF NEUROSCIENCE

Search-and-replace genome editing without double-strand breaks or donor DNA

(2019) Andrew V. Anzalone et al.   NATURE

Increased Ca2+ signaling in NRXN1α+/− neurons derived from ASD induced pluripotent stem cells

(2019) Sahar Avazzadeh et al.   Molecular Autism

Sex-dependent effects of Cacna1c haploinsufficiency on behavioral inhibition evoked by conspecific alarm signals in rats

(2019) Markus Wöhr et al.   PROGRESS IN NEURO-PSYCHOPHARMACOLOGY & BIOLOGICAL PSYCHIATRY

Dual Molecular Effects of Dominant RORA Mutations Cause Two Variants of Syndromic Intellectual Disability with Either Autism or Cerebellar Ataxia

(2018) Claire Guissart et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Estimation of clinical trial success rates and related parameters

(2018) Chi Heem Wong et al.   BIOSTATISTICS

Embryonic exposure to valproic acid affects the histaminergic system and the social behaviour of adult zebrafish (Danio rerio )

(2018) Diego Baronio et al.   BRITISH JOURNAL OF PHARMACOLOGY

Transcriptome Engineering with RNA-Targeting Type VI-D CRISPR Effectors

(2018) Silvana Konermann et al.   CELL

Altered Neocortical Gene Expression, Brain Overgrowth and Functional Over-Connectivity in Chd8 Haploinsufficient Mice

(2018) Philipp Suetterlin et al.   CEREBRAL CORTEX

A Forward Genetic Screen in Zebrafish Identifies the G-Protein-Coupled Receptor CaSR as a Modulator of Sensorimotor Decision Making

(2018) Roshan A. Jain et al.   CURRENT BIOLOGY

Dual Requirement of CHD8 for Chromatin Landscape Establishment and Histone Methyltransferase Recruitment to Promote CNS Myelination and Repair

(2018) Chuntao Zhao et al.   DEVELOPMENTAL CELL

Progress in the genetics of autism spectrum disorder

(2018) Marc Woodbury-Smith et al.   DEVELOPMENTAL MEDICINE AND CHILD NEUROLOGY

Cacna1c haploinsufficiency leads to pro-social 50-kHz ultrasonic communication deficits in rats

(2018) Theresa M. Kisko et al.   Disease Models & Mechanisms

Zebrafish models of autism spectrum disorder

(2018) Daria A. Meshalkina et al.   EXPERIMENTAL NEUROLOGY

Rare RELN variants affect Reelin-DAB1 signal transduction in autism spectrum disorder

(2018) Sandra M. Sánchez-Sánchez et al.   HUMAN MUTATION

Altered TAOK2 activity causes autism-related neurodevelopmental and cognitive abnormalities through RhoA signaling

(2018) Melanie Richter et al.   MOLECULAR PSYCHIATRY

Purkinje cells derived from TSC patients display hypoexcitability and synaptic deficits associated with reduced FMRP levels and reversed by rapamycin

(2018) Maria Sundberg et al.   MOLECULAR PSYCHIATRY

p53 inhibits CRISPR–Cas9 engineering in human pluripotent stem cells

(2018) Robert J. Ihry et al.   NATURE MEDICINE

CRISPR–Cas9 genome editing induces a p53-mediated DNA damage response

(2018) Emma Haapaniemi et al.   NATURE MEDICINE

Intermittent fasting uncovers and rescues cognitive phenotypes in PTEN neuronal haploinsufficient mice

(2018) J. V. Cabral-Costa et al.   Scientific Reports

CRISPR/Cas9-induced shank3b mutant zebrafish display autism-like behaviors

(2018) Chun-xue Liu et al.   Molecular Autism

Single-cell trajectory analysis of human homogenous neurons carrying a rare RELN variant

(2018) Yuko Arioka et al.   Translational Psychiatry

Hyperactivity and Hypermotivation Associated With Increased Striatal mGluR1 Signaling in a Shank2 Rat Model of Autism

(2018) Meera E. Modi et al.   Frontiers in Molecular Neuroscience

Altered auditory processing, filtering, and reactivity in theCntnap2knockout rat model for neurodevelopmental disorders

(2018) Kaela E. Scott et al.   JOURNAL OF NEUROSCIENCE

Evolved Cas9 variants with broad PAM compatibility and high DNA specificity

(2018) Johnny H. Hu et al.   NATURE

Sexually dimorphic behavior, neuronal activity, and gene expression in Chd8-mutant mice

(2018) Hwajin Jung et al.   NATURE NEUROSCIENCE

Plausibility of the zebrafish embryos/larvae as an alternative animal model for autism: A comparison study of transcriptome changes

(2018) Sangwoo Lee et al.   PLoS One

Zebrafish Models of Neurodevelopmental Disorders: Past, Present, and Future

(2018) Catalina Sakai et al.   Frontiers in Molecular Neuroscience

De novo Mutations (DNMs) in Autism Spectrum Disorder (ASD): Pathway and Network Analysis

(2018) Aitana Alonso-Gonzalez et al.   Frontiers in Genetics

Early Correction of N-Methyl-D-Aspartate Receptor Function Improves Autistic-like Social Behaviors in Adult Shank2−/− Mice

(2018) Changuk Chung et al.   BIOLOGICAL PSYCHIATRY

Chd2 Is Necessary for Neural Circuit Development and Long-Term Memory

(2018) Young J. Kim et al.   NEURON

Neonatal Tbr1 Dosage Controls Cortical Layer 6 Connectivity

(2018) Siavash Fazel Darbandi et al.   NEURON

Convergent microstructural brain changes across genetic models of autism spectrum disorder—A pilot study

(2018) Paul A. Rowley et al.   PSYCHIATRY RESEARCH-NEUROIMAGING

Larval zebrafish model for studying the effects of valproic acid on neurodevelopment: An approach towards modeling autism

(2018) Shubham Dwivedi et al.   JOURNAL OF PHARMACOLOGICAL AND TOXICOLOGICAL METHODS

Developmental protein kinase C hyper-activation results in microcephaly and behavioral abnormalities in zebrafish

(2018) Taian Liu et al.   Translational Psychiatry

Complete Disruption of Autism-Susceptibility Genes by Gene Editing Predominantly Reduces Functional Connectivity of Isogenic Human Neurons

(2018) Eric Deneault et al.   Stem Cell Reports

Arid1b Haploinsufficiency Causes Abnormal Brain Gene Expression and Autism-Related Behaviors in Mice

(2017) Mihiro Shibutani et al.   INTERNATIONAL JOURNAL OF MOLECULAR SCIENCES

What Is the Male-to-Female Ratio in Autism Spectrum Disorder? A Systematic Review and Meta-Analysis

(2017) Rachel Loomes et al.   JOURNAL OF THE AMERICAN ACADEMY OF CHILD AND ADOLESCENT PSYCHIATRY

Adult zebrafish in CNS disease modeling: a tank that's half-full, not half-empty, and still filling

(2017) Darya A Meshalkina et al.   LAB ANIMAL

PREPARE: guidelines for planning animal research and testing

(2017) Adrian J Smith et al.   LABORATORY ANIMALS

A Upf3b-mutant mouse model with behavioral and neurogenesis defects

(2017) L Huang et al.   MOLECULAR PSYCHIATRY

Stem cell-derived neurons from autistic individuals with SHANK3 mutation show morphogenetic abnormalities during early development

(2017) A Kathuria et al.   MOLECULAR PSYCHIATRY

RNA targeting with CRISPR–Cas13

(2017) Omar O. Abudayyeh et al.   NATURE

Programmable base editing of A•T to G•C in genomic DNA without DNA cleavage

(2017) Nicole M. Gaudelli et al.   NATURE

Disruption of the ATXN1–CIC complex causes a spectrum of neurobehavioral phenotypes in mice and humans

(2017) Hsiang-Chih Lu et al.   NATURE GENETICS

Germline Chd8 haploinsufficiency alters brain development in mouse

(2017) Andrea L Gompers et al.   NATURE NEUROSCIENCE

Linking Neurons to Network Function and Behavior by Two-Photon Holographic Optogenetics and Volumetric Imaging

(2017) Marco dal Maschio et al.   NEURON

Enhancing inhibitory synaptic function reverses spatial memory deficits in Shank2 mutant mice

(2017) Chae-Seok Lim et al.   NEUROPHARMACOLOGY

Generation and characterization of Kctd15 mutations in zebrafish

(2017) Alison Heffer et al.   PLoS One

Sex-specific gene–environment interactions underlying ASD-like behaviors

(2017) Sara M. Schaafsma et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Modulation of prefrontal cortex excitation/inhibition balance rescues social behavior in CNTNAP2 -deficient mice

(2017) Aslihan Selimbeyoglu et al.   Science Translational Medicine

Sleep/Wake Physiology and Quantitative Electroencephalogram Analysis of the Neuroligin-3 Knockout Rat Model of Autism Spectrum Disorder

(2017) Alexia M Thomas et al.   SLEEP

Fragile X Mental Retardation-1 Knockout Zebrafish Shows Precocious Development in Social Behavior

(2017) Yao-Ju Wu et al.   Zebrafish

Chd8 Mutation Leads to Autistic-like Behaviors and Impaired Striatal Circuits

(2017) Randall J. Platt et al.   Cell Reports

Guidelines for morpholino use in zebrafish

(2017) Didier Y. R. Stainier et al.   PLoS Genetics

MYT1L mutations cause intellectual disability and variable obesity by dysregulating gene expression and development of the neuroendocrine hypothalamus

(2017) Patricia Blanchet et al.   PLoS Genetics

Oxytocin improves behavioral and electrophysiological deficits in a novel Shank3-deficient rat

(2017) Hala Harony-Nicolas et al.   eLife

Arid1b haploinsufficient mice reveal neuropsychiatric phenotypes and reversible causes of growth impairment

(2017) Cemre Celen et al.   eLife

CRISPR/Cas9-mediated heterozygous knockout of the autism gene CHD8 and characterization of its transcriptional networks in cerebral organoids derived from iPS cells

(2017) Ping Wang et al.   Molecular Autism

Zebrafish knockout of Down syndrome gene, DYRK1A, shows social impairments relevant to autism

(2017) Oc-Hee Kim et al.   Molecular Autism

Environmental risk factors for autism: an evidence-based review of systematic reviews and meta-analyses

(2017) Amirhossein Modabbernia et al.   Molecular Autism

Altered neurite morphology and cholinergic function of induced pluripotent stem cell-derived neurons from a patient with Kleefstra syndrome and autism

(2017) J Nagy et al.   Translational Psychiatry

Editorial: Autism Spectrum Disorders: Developmental Trajectories, Neurobiological Basis, Treatment Update

(2017) Roberto Canitano et al.   Frontiers in Psychiatry

Sleep/Wake Physiology and Quantitative Electroencephalogram Analysis of the Neuroligin-3 Knockout Rat Model of Autism Spectrum Disorder

(2017) Alexia M Thomas et al.   SLEEP

Peripheral Mechanosensory Neuron Dysfunction Underlies Tactile and Behavioral Deficits in Mouse Models of ASDs

(2016) Lauren L. Orefice et al.   CELL

Biology and Applications of CRISPR Systems: Harnessing Nature’s Toolbox for Genome Engineering

(2016) Addison V. Wright et al.   CELL

Rodent models in neuroscience research: is it a rat race?

(2016) Bart Ellenbroek et al.   Disease Models & Mechanisms

Characterization of Rett Syndrome-like phenotypes in Mecp2-knockout rats

(2016) Yang Wu et al.   Journal of Neurodevelopmental Disorders

QRFP and Its Receptors Regulate Locomotor Activity and Sleep in Zebrafish

(2016) A. Chen et al.   JOURNAL OF NEUROSCIENCE

Cerebellar Shank2 Regulates Excitatory Synapse Density, Motor Coordination, and Specific Repetitive and Anxiety-Like Behaviors

(2016) S. Ha et al.   JOURNAL OF NEUROSCIENCE

Sexual divergence in microtubule function: the novel intranasal microtubule targeting SKIP normalizes axonal transport and enhances memory

(2016) N Amram et al.   MOLECULAR PSYCHIATRY

CHD8 haploinsufficiency results in autistic-like phenotypes in mice

(2016) Yuta Katayama et al.   NATURE

Chd8 mediates cortical neurogenesis via transcriptional regulation of cell cycle and Wnt signaling

(2016) Omer Durak et al.   NATURE NEUROSCIENCE

Induced pluripotent stem cell technology: a decade of progress

(2016) Yanhong Shi et al.   NATURE REVIEWS DRUG DISCOVERY

Estrogens Suppress a Behavioral Phenotype in Zebrafish Mutants of the Autism Risk Gene, CNTNAP2

(2016) Ellen J. Hoffman et al.   NEURON

Alternative Animal and Non-Animal Models for Drug Discovery and Development: Bonus or Burden?

(2016) Irlan Almeida Freires et al.   PHARMACEUTICAL RESEARCH

A Spontaneous Missense Mutation in Branched Chain Keto Acid Dehydrogenase Kinase in the Rat Affects Both the Central and Peripheral Nervous Systems

(2016) J. Samuel Zigler et al.   PLoS One

Cntnap2 Knockout Rats and Mice Exhibit Epileptiform Activity and Abnormal Sleep–Wake Physiology

(2016) Alexia M. Thomas et al.   SLEEP

On the Origin of CRISPR-Cas Technology: From Prokaryotes to Mammals

(2016) Francisco J.M. Mojica et al.   TRENDS IN MICROBIOLOGY

Histone methyltransferase Ash1L mediates activity-dependent repression of neurexin-1α

(2016) Τao Zhu et al.   Scientific Reports

Impaired Mitochondrial Dynamics and Mitophagy in Neuronal Models of Tuberous Sclerosis Complex

(2016) Darius Ebrahimi-Fakhari et al.   Cell Reports

Human Pluripotent Stem Cell-derived Cortical Neurons for High Throughput Medication Screening in Autism: A Proof of Concept Study in SHANK3 Haploinsufficiency Syndrome

(2016) Hélène Darville et al.   EBioMedicine

Cntnap2 Knockout Rats and Mice Exhibit Epileptiform Activity and Abnormal Sleep–Wake Physiology

(2016) Alexia M. Thomas et al.   SLEEP

Transcriptional enhancement of Smn levels in motoneurons is crucial for proper axon morphology in zebrafish

(2016) Zoltán Spiró et al.   Scientific Reports

Gain-of-function defects of astrocytic Kir4.1 channels in children with autism spectrum disorders and epilepsy

(2016) Federico Sicca et al.   Scientific Reports

Phenotypic characterization of nonsocial behavioral impairment in neurexin 1α knockout rats.

(2015) Frederic Esclassan et al.   BEHAVIORAL NEUROSCIENCE

The Histone Methyltransferase Gene Absent, Small, or Homeotic Discs-1 Like Is Required for Normal Hox Gene Expression and Fertility in Mice1

(2015) Michelle L. Brinkmeier et al.   BIOLOGY OF REPRODUCTION

Necessary Conditions for Morally Responsible Animal Research

(2015) DAVID DeGRAZIA et al.   CAMBRIDGE QUARTERLY OF HEALTHCARE ETHICS

Neurobeachin Is Required Postsynaptically for Electrical and Chemical Synapse Formation

(2015) Adam C. Miller et al.   CURRENT BIOLOGY

Collybistin binds and inhibits mTORC1 signaling: a potential novel mechanism contributing to intellectual disability and autism

(2015) Camila Oliveira Freitas Machado et al.   EUROPEAN JOURNAL OF HUMAN GENETICS

Decreased aggression and increased repetitive behavior inPtenhaploinsufficient mice

(2015) A. E. Clipperton-Allen et al.   GENES BRAIN AND BEHAVIOR

Copy number variants in patients with intellectual disability affect the regulation of ARX transcription factor gene

(2015) Minaka Ishibashi et al.   HUMAN GENETICS

Two knockdown models of the autism genes SYNGAP1 and SHANK3 in zebrafish produce similar behavioral phenotypes associated with embryonic disruptions of brain morphogenesis

(2015) Robert A. Kozol et al.   HUMAN MOLECULAR GENETICS

Kabuki syndrome genesKMT2DandKDM6A: functional analyses demonstrate critical roles in craniofacial, heart and brain development

(2015) Peter M. Van Laarhoven et al.   HUMAN MOLECULAR GENETICS

RAP1-mediated MEK/ERK pathway defects in Kabuki syndrome

(2015) Nina Bögershausen et al.   JOURNAL OF CLINICAL INVESTIGATION

Hyperactivity of Newborn Pten Knock-out Neurons Results from Increased Excitatory Synaptic Drive

(2015) M. R. Williams et al.   JOURNAL OF NEUROSCIENCE

Altered neuronal network and rescue in a human MECP2 duplication model

(2015) S Nageshappa et al.   MOLECULAR PSYCHIATRY

Genetic and functional analyses demonstrate a role for abnormal glycinergic signaling in autism

(2015) M Pilorge et al.   MOLECULAR PSYCHIATRY

Loss of δ-catenin function in severe autism

(2015) Tychele N. Turner et al.   NATURE

CRISPRscan: designing highly efficient sgRNAs for CRISPR-Cas9 targeting in vivo

(2015) Miguel A Moreno-Mateos et al.   NATURE METHODS

Degraded neural and behavioral processing of speech sounds in a rat model of Rett syndrome

(2015) Crystal T. Engineer et al.   NEUROBIOLOGY OF DISEASE

Restoration of Normal Cerebral Oxygen Consumption with Rapamycin Treatment in a Rat Model of Autism–Tuberous Sclerosis

(2015) Oak Z. Chi et al.   NEUROMOLECULAR MEDICINE

Autistic-Like Traits and Cerebellar Dysfunction in Purkinje Cell PTEN Knock-Out Mice

(2015) Dario Cupolillo et al.   NEUROPSYCHOPHARMACOLOGY

Abnormal neuronal morphology and neurochemistry in the auditory brainstem of Fmr1 knockout rats

(2015) K. Ruby et al.   NEUROSCIENCE

Prenatal factors associated with autism spectrum disorder (ASD)

(2015) A. Ornoy et al.   REPRODUCTIVE TOXICOLOGY

A prudent path forward for genomic engineering and germline gene modification

(2015) D. Baltimore et al.   SCIENCE

Rationally engineered Cas9 nucleases with improved specificity

(2015) I. M. Slaymaker et al.   SCIENCE

Exogenous and evoked oxytocin restores social behavior in the Cntnap2 mouse model of autism

(2015) Olga Peñagarikano et al.   Science Translational Medicine

Methyl-CpG Binding Protein 2 (Mecp2) Regulates Sensory Function Through Sema5b and Robo2

(2015) Wan Y. Leong et al.   Frontiers in Cellular Neuroscience

Trans-synaptic zinc mobilization improves social interaction in two mouse models of autism through NMDAR activation

(2015) Eun-Jae Lee et al.   Nature Communications

The Parvalbumin/Somatostatin Ratio Is Increased in Pten Mutant Mice and by Human PTEN ASD Alleles

(2015) Daniel Vogt et al.   Cell Reports

Advancing biology through a deeper understanding of zebrafish ecology and evolution

(2015) David M Parichy   eLife

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

ZNF804A Transcriptional Networks in Differentiating Neurons Derived from Induced Pluripotent Stem Cells of Human Origin

(2015) Jian Chen et al.   PLoS One

Fmr1 and Nlgn3 knockout rats: Novel tools for investigating autism spectrum disorders.

(2014) Shannon M. Hamilton et al.   BEHAVIORAL NEUROSCIENCE

Development and Applications of CRISPR-Cas9 for Genome Engineering

(2014) Patrick D. Hsu et al.   CELL

Disruptive CHD8 Mutations Define a Subtype of Autism Early in Development

(2014) Raphael Bernier et al.   CELL

Whole-Genome Sequencing Analysis Reveals High Specificity of CRISPR/Cas9 and TALEN-Based Genome Editing in Human iPSCs

(2014) Cory Smith et al.   Cell Stem Cell

Methylphenidate improves learning impairments and hyperthermia-induced seizures caused by anScn1amutation

(2014) Iori Ohmori et al.   EPILEPSIA

In vivo detection of reduced Purkinje cell fibers with diffusion MRI tractography in children with autistic spectrum disorders

(2014) Jeong-Won Jeong et al.   Frontiers in Human Neuroscience

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

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

The contribution of de novo coding mutations to autism spectrum disorder

(2014) Ivan Iossifov et al.   NATURE

Tbr1 haploinsufficiency impairs amygdalar axonal projections and results in cognitive abnormality

(2014) Tzyy-Nan Huang et al.   NATURE NEUROSCIENCE

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

Anxiety, hyperactivity and stereotypy in a zebrafish model of fragile X syndrome and autism spectrum disorder

(2014) Lily Kim et al.   PROGRESS IN NEURO-PSYCHOPHARMACOLOGY & BIOLOGICAL PSYCHIATRY

Zebrafish models of human motor neuron diseases: Advantages and limitations

(2014) Patrick J. Babin et al.   PROGRESS IN NEUROBIOLOGY

The new frontier of genome engineering with CRISPR-Cas9

(2014) J. A. Doudna et al.   SCIENCE

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

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

Induction of Social Behavior in Zebrafish: Live Versus Computer Animated Fish as Stimuli

(2014) Meiying Qin et al.   Zebrafish

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

(2014) Yuan Tian et al.   Genome Medicine

Involvement of Synaptic Genes in the Pathogenesis of Autism Spectrum Disorders: The Case of Synapsins

(2014) Silvia Giovedí et al.   Frontiers in Pediatrics

De Novo Loss-of-Function Mutations in CHD2 Cause a Fever-Sensitive Myoclonic Epileptic Encephalopathy Sharing Features with Dravet Syndrome

(2013) Arvid Suls et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Decreased Reelin Expression and Organophosphate Pesticide Exposure Alters Mouse Behaviour and Brain Morphology

(2013) Brian R. Mullen et al.   ASN Neuro

Behavioural methods used in rodent models of autism spectrum disorders: Current standards and new developments

(2013) Markus Wöhr et al.   BEHAVIOURAL BRAIN RESEARCH

Maturation of shoaling in two zebrafish strains: A behavioral and neurochemical analysis

(2013) Samantha Mahabir et al.   BEHAVIOURAL BRAIN RESEARCH

New approach for fish breeding by chemical mutagenesis: establishment of TILLING method in fugu (Takifugu rubripes) with ENU mutagenesis

(2013) Miwa Kuroyanagi et al.   BMC GENOMICS

Repurposing CRISPR as an RNA-Guided Platform for Sequence-Specific Control of Gene Expression

(2013) Lei S. Qi et al.   CELL

Defining the Role of Cerebellar Purkinje Cells in Autism Spectrum Disorders

(2013) Anamaria Sudarov   CEREBELLUM

Cloacal exstrophy with extensive Chiari II malformation: case report and review of the literature

(2013) Obed M. Nyarenchi et al.   CHILDS NERVOUS SYSTEM

Transgenic tools to characterize neuronal properties of discrete populations of zebrafish neurons

(2013) C. Satou et al.   DEVELOPMENT

Genome editing using artificial site-specific nucleases in zebrafish

(2013) Yu Hisano et al.   DEVELOPMENT GROWTH & DIFFERENTIATION

Pharmacotherapy for the Core Symptoms in Autistic Disorder: Current Status of the Research

(2013) Cristan Farmer et al.   DRUGS

Analysis of off-target effects of CRISPR/Cas-derived RNA-guided endonucleases and nickases

(2013) S. W. Cho et al.   GENOME RESEARCH

Histone Methyltransferase Ash1l Suppresses Interleukin-6 Production and Inflammatory Autoimmune Diseases by Inducing the Ubiquitin-Editing Enzyme A20

(2013) Meng Xia et al.   IMMUNITY

Calcium influx through L-type CaV1.2 Ca2+ channels regulates mandibular development

(2013) Kapil V. Ramachandran et al.   JOURNAL OF CLINICAL INVESTIGATION

Modeling Transformations of Neurodevelopmental Sequences across Mammalian Species

(2013) A. D. Workman et al.   JOURNAL OF NEUROSCIENCE

SHANK3 and IGF1 restore synaptic deficits in neurons from 22q13 deletion syndrome patients

(2013) Aleksandr Shcheglovitov et al.   NATURE

The zebrafish reference genome sequence and its relationship to the human genome

(2013) Kerstin Howe et al.   NATURE

Efficient genome editing in zebrafish using a CRISPR-Cas system

(2013) Woong Y Hwang et al.   NATURE BIOTECHNOLOGY

Heritable gene targeting in the mouse and rat using a CRISPR-Cas system

(2013) Dali Li et al.   NATURE BIOTECHNOLOGY

Whole-brain functional imaging at cellular resolution using light-sheet microscopy

(2013) Misha B Ahrens et al.   NATURE METHODS

RNA-guided gene activation by CRISPR-Cas9–based transcription factors

(2013) Pablo Perez-Pinera et al.   NATURE METHODS

Heritable genome editing in C. elegans via a CRISPR-Cas9 system

(2013) Ari E Friedland et al.   NATURE METHODS

Functional Impacts of NRXN1 Knockdown on Neurodevelopment in Stem Cell Models

(2013) Liyun Zeng et al.   PLoS One

Hippocampal Neuroligin-2 Overexpression Leads to Reduced Aggression and Inhibited Novelty Reactivity in Rats

(2013) Christine Kohl et al.   PLoS One

The first mecp2-null zebrafish model shows altered motor behaviors

(2013) Thomas Pietri et al.   Frontiers in Neural Circuits

Toward developmental models of psychiatric disorders in zebrafish

(2013) William H. J. Norton   Frontiers in Neural Circuits

Highly Efficient Targeted Mutagenesis of Drosophila with the CRISPR/Cas9 System

(2013) Andrew R. Bassett et al.   Cell Reports

Function and Regulation of AUTS2, a Gene Implicated in Autism and Human Evolution

(2013) Nir Oksenberg et al.   PLoS Genetics

RNA-programmed genome editing in human cells

(2013) Martin Jinek et al.   eLife

SFARI Gene 2.0: a community-driven knowledgebase for the autism spectrum disorders (ASDs)

(2013) Brett S Abrahams et al.   Molecular Autism

Neurodevelopmental disorders: mechanisms and boundary definitions from genomes, interactomes and proteomes

(2013) A P Mullin et al.   Translational Psychiatry

Differentiation from human pluripotent stem cells of cortical neurons of the superficial layers amenable to psychiatric disease modeling and high-throughput drug screening

(2013) C Boissart et al.   Translational Psychiatry

The BTBR T+tf/J mouse model for autism spectrum disorders–in search of biomarkers

(2012) Ksenia Z. Meyza et al.   BEHAVIOURAL BRAIN RESEARCH

Chondroitin Fragments Are Odorants that Trigger Fear Behavior in Fish

(2012) Ajay S. Mathuru et al.   CURRENT BIOLOGY

Touch responsiveness in zebrafish requires voltage-gated calcium channel 2.1b

(2012) Sean E. Low et al.   JOURNAL OF NEUROPHYSIOLOGY

Ontogeny of classical and operant learning behaviors in zebrafish

(2012) A. Valente et al.   LEARNING & MEMORY

Modeling psychiatric disorders at the cellular and network levels

(2012) K J Brennand et al.   MOLECULAR PSYCHIATRY

Autistic-like social behaviour in Shank2-mutant mice improved by restoring NMDA receptor function

(2012) Hyejung Won et al.   NATURE

TALENs: a widely applicable technology for targeted genome editing

(2012) J. Keith Joung et al.   NATURE REVIEWS MOLECULAR CELL BIOLOGY

The dorsal pallium in zebrafish, Danio rerio (Cyprinidae, Teleostei)

(2011) Thomas Mueller et al.   BRAIN RESEARCH

Absence of CNTNAP2 Leads to Epilepsy, Neuronal Migration Abnormalities, and Core Autism-Related Deficits

(2011) Olga Peñagarikano et al.   CELL

Transgenic zebrafish expressing green fluorescent protein in dopaminergic neurons of the ventral diencephalon

(2011) Yanwei Xi et al.   DEVELOPMENTAL DYNAMICS

Genome Engineering With Zinc-Finger Nucleases

(2011) Dana Carroll   GENETICS

Heritable gene targeting in zebrafish using customized TALENs

(2011) Peng Huang et al.   NATURE BIOTECHNOLOGY

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

(2011) Sergiu P Paşca et al.   NATURE MEDICINE

Zebrafish neurotransmitter systems as potential pharmacological and toxicological targets

(2011) E.P. Rico et al.   NEUROTOXICOLOGY AND TERATOLOGY

Modeling Autistic Features in Animals

(2011) PAUL H. PATTERSON   PEDIATRIC RESEARCH

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

The role of dopaminergic signalling during larval zebrafish brain development: a tool for investigating the developmental basis of neuropsychiatric disorders

(2011) Bruno Rezende Souza et al.   REVIEWS IN THE NEUROSCIENCES

Genetics of autism spectrum disorders

(2011) Daniel H. Geschwind   TRENDS IN COGNITIVE SCIENCES

Somatosensory and sensorimotor consequences associated with the heterozygous disruption of the autism candidate gene, Gabrb3

(2010) Timothy M. DeLorey et al.   BEHAVIOURAL BRAIN RESEARCH

Proneural gene-linked neurogenesis in zebrafish cerebellum

(2010) Shuichi Kani et al.   DEVELOPMENTAL BIOLOGY

Targeted Genome Modification in Mice Using Zinc-Finger Nucleases

(2010) I. D. Carbery et al.   GENETICS

Targeting DNA Double-Strand Breaks with TAL Effector Nucleases

(2010) M. Christian et al.   GENETICS

Organization of Myelinated Axons by Caspr and Caspr2 Requires the Cytoskeletal Adapter Protein 4.1B

(2010) I. Horresh et al.   JOURNAL OF NEUROSCIENCE

Behavioural phenotyping assays for mouse models of autism

(2010) Jill L. Silverman et al.   NATURE REVIEWS NEUROSCIENCE

DNA resection in eukaryotes: deciding how to fix the break

(2010) Pablo Huertas   NATURE STRUCTURAL & MOLECULAR BIOLOGY

Neuronal voltage-gated ion channels are genetic modifiers of generalized epilepsy with febrile seizures plus

(2010) Nicole A. Hawkins et al.   NEUROBIOLOGY OF DISEASE

Improving Bioscience Research Reporting: The ARRIVE Guidelines for Reporting Animal Research

(2010) Carol Kilkenny et al.   PLOS BIOLOGY

Characteristics and Concordance of Autism Spectrum Disorders Among 277 Twin Pairs

(2009) Rebecca E. Rosenberg et al.   ARCHIVES OF PEDIATRICS & ADOLESCENT MEDICINE

An Evolutionary Interpretation of Teleostean Forebrain Anatomy

(2009) Thomas Mueller et al.   BRAIN BEHAVIOR AND EVOLUTION

The autism susceptibility gene met regulates zebrafish cerebellar development and facial motor neuron migration

(2009) Gina E. Elsen et al.   DEVELOPMENTAL BIOLOGY

Anatomy of zebrafish cerebellum and screen for mutations affecting its development

(2009) Young-Ki Bae et al.   DEVELOPMENTAL BIOLOGY

Using zebrafish to assess the impact of drugs on neural development and function

(2009) Su Guo   Expert Opinion on Drug Discovery

Pharmacological Inhibition of mTORC1 Suppresses Anatomical, Cellular, and Behavioral Abnormalities in Neural-Specific Pten Knock-Out Mice

(2009) J. Zhou et al.   JOURNAL OF NEUROSCIENCE

Environmental risk factors for autism: Do they help cause de novo genetic mutations that contribute to the disorder?

(2009) Dennis K. Kinney et al.   MEDICAL HYPOTHESES

Efficient targeting of expressed and silent genes in human ESCs and iPSCs using zinc-finger nucleases

(2009) Dirk Hockemeyer et al.   NATURE BIOTECHNOLOGY

CHD8 suppresses p53-mediated apoptosis through histone H1 recruitment during early embryogenesis

(2009) Masaaki Nishiyama et al.   NATURE CELL BIOLOGY

Role of chromodomain helicase DNA-binding protein 2 in DNA damage response signaling and tumorigenesis

(2009) P Nagarajan et al.   ONCOGENE

The transmembrane inner ear (Tmie) protein is essential for normal hearing and balance in the zebrafish

(2009) Michelle R. Gleason et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

A Primer for Morpholino Use in Zebrafish

(2009) Brent R. Bill et al.   Zebrafish

Controlling morpholino experiments: don't stop making antisense

(2008) J. S. Eisen et al.   DEVELOPMENT

Axonal projections originating from raphe serotonergic neurons in the developing and adult zebrafish,Danio rerio, using transgenics to visualize raphe-specificpet1expression

(2008) Christina Lillesaar et al.   JOURNAL OF COMPARATIVE NEUROLOGY

Vesicular Glutamate Transporter 3 Is Required for Synaptic Transmission in Zebrafish Hair Cells

(2008) N. Obholzer et al.   JOURNAL OF NEUROSCIENCE

Heritable targeted gene disruption in zebrafish using designed zinc-finger nucleases

(2008) Yannick Doyon et al.   NATURE BIOTECHNOLOGY

Alarm substance induced behavioral responses in zebrafish (Danio rerio)

(2007) Natasha Speedie et al.   BEHAVIOURAL BRAIN RESEARCH

Visualization of monoaminergic neurons and neurotoxicity of MPTP in live transgenic zebrafish

(2007) Lu Wen et al.   DEVELOPMENTAL BIOLOGY