Using C. elegans to Decipher the Cellular and Molecular Mechanisms Underlying Neurodevelopmental Disorders

SHANK1 Deletions in Males with Autism Spectrum Disorder

(2012) Daisuke Sato et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Wnt Signaling Regulates Acetylcholine Receptor Translocation and Synaptic Plasticity in the Adult Nervous System

(2012) Michael Jensen et al.   CELL

Studies on Aplysia neurons suggest treatments for chronic human disorders

(2012) Thomas W. Abrams   CURRENT BIOLOGY

Inhibition of tau aggregation in a novel Caenorhabditis elegans model of tauopathy mitigates proteotoxicity

(2012) Chronis Fatouros et al.   HUMAN MOLECULAR GENETICS

The doublecortin-related genezyg-8is a microtubule organizer inCaenorhabditis elegansneurons

(2012) Jean-Michel Bellanger et al.   JOURNAL OF CELL SCIENCE

Mutations inDYNC1H1cause severe intellectual disability with neuronal migration defects

(2012) Marjolein H Willemsen et al.   JOURNAL OF MEDICAL GENETICS

Laterally Orienting C. elegans Using Geometry at Microscale for High-Throughput Visual Screens in Neurodegeneration and Neuronal Development Studies

(2012) Ivan de Carlos Cáceres et al.   PLoS One

Functional Phenotypic Rescue of Caenorhabditis elegans Neuroligin-Deficient Mutants by the Human and Rat NLGN1 Genes

(2012) Fernando Calahorro et al.   PLoS One

Optical Silencing of C. elegans Cells with Arch Proton Pump

(2012) Ayako Okazaki et al.   PLoS One

Cross-Species Functional Genomic Analysis Identifies Resistance Genes of the Histone Deacetylase Inhibitor Valproic Acid

(2012) Rakel Brendsdal Forthun et al.   PLoS One

Photo-inducible cell ablation in Caenorhabditis elegans using the genetically encoded singlet oxygen generating protein miniSOG

(2012) Y. B. Qi et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

DISC1: Structure, Function, and Therapeutic Potential for Major Mental Illness

(2011) Dinesh C. Soares et al.   ACS Chemical Neuroscience

Human Mutations in NDE1 Cause Extreme Microcephaly with Lissencephaly

(2011) Fowzan S. Alkuraya et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Effects of a selective Y2R antagonist, JNJ-31020028, on nicotine abstinence-related social anxiety-like behavior, neuropeptide Y and corticotropin releasing factor mRNA levels in the novelty-seeking phenotype

(2011) Cigdem Aydin et al.   BEHAVIOURAL BRAIN RESEARCH

TRPM Channels Modulate Epileptic-like Convulsions via Systemic Ion Homeostasis

(2011) Tamara M. Stawicki et al.   CURRENT BIOLOGY

The Autism Candidate Gene Neurobeachin Encodes a Scaffolding Protein Implicated in Membrane Trafficking and Signaling

(2011) K. Volders et al.   CURRENT MOLECULAR MEDICINE

Drosophila modeling of heritable neurodevelopmental disorders

(2011) Cheryl L Gatto et al.   CURRENT OPINION IN NEUROBIOLOGY

Cilia in the nervous system: linking cilia function and neurodevelopmental disorders

(2011) Ji E Lee et al.   CURRENT OPINION IN NEUROLOGY

Identification of the AFD neuron as the site of action of the CREB protein in Caenorhabditis elegans thermotaxis

(2011) Yukuo Nishida et al.   EMBO REPORTS

Neuron-specific proteotoxicity of mutant ataxin-3 in C. elegans : rescue by the DAF-16 and HSF-1 pathways

(2011) Andreia Teixeira-Castro et al.   HUMAN MOLECULAR GENETICS

Caenorhabditis elegans as an experimental tool for the study of complex neurological diseases: Parkinson’s disease, Alzheimer’s disease and autism spectrum disorder

(2011) Fernando Calahorro et al.   INVERTEBRATE NEUROSCIENCE

The Presynaptic Dense Projection of the Caenorhabiditis elegans Cholinergic Neuromuscular Junction Localizes Synaptic Vesicles at the Active Zone through SYD-2/Liprin and UNC-10/RIM-Dependent Interactions

(2011) C. Stigloher et al.   JOURNAL OF NEUROSCIENCE

NrCAM Deletion Causes Topographic Mistargeting of Thalamocortical Axons to the Visual Cortex and Disrupts Visual Acuity

(2011) G. P. Demyanenko et al.   JOURNAL OF NEUROSCIENCE

aPKC phosphorylates NuMA-related LIN-5 to position the mitotic spindle during asymmetric division

(2011) Matilde Galli et al.   NATURE CELL BIOLOGY

Small-molecule proteostasis regulators for protein conformational diseases

(2011) Barbara Calamini et al.   Nature Chemical Biology

Optogenetic manipulation of neural activity in freely moving Caenorhabditis elegans

(2011) Andrew M Leifer et al.   NATURE METHODS

OrthoList: A Compendium of C. elegans Genes with Human Orthologs

(2011) Daniel D. Shaye et al.   PLoS One

Disrupted-in-Schizophrenia 1-mediated axon guidance involves TRIO-RAC-PAK small GTPase pathway signaling

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

Caenorhabditis elegans aristaless/Arx gene alr-1 restricts variable gene expression

(2011) I. Topalidou et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

The neural circuits and sensory channels mediating harsh touch sensation in Caenorhabditis elegans

(2011) Wei Li et al.   Nature Communications

Optogenetic analysis of synaptic transmission in the central nervous system of the nematode Caenorhabditis elegans

(2011) Theodore H. Lindsay et al.   Nature Communications

Human ASPM participates in spindle organisation, spindle orientation and cytokinesis

(2010) Julie Higgins et al.   BMC CELL BIOLOGY

The X-linked mental retardation gene PHF8 is a histone demethylase involved in neuronal differentiation

(2010) Jihui Qiu et al.   CELL RESEARCH

ATRX Partners with Cohesin and MeCP2 and Contributes to Developmental Silencing of Imprinted Genes in the Brain

(2010) Kristin D. Kernohan et al.   DEVELOPMENTAL CELL

Neuroligin-deficient mutants of C. elegans have sensory processing deficits and are hypersensitive to oxidative stress and mercury toxicity

(2010) J. W. Hunter et al.   Disease Models & Mechanisms

Polymorphisms in CACNA1E and Camk2d are associated with seizure susceptibility of Sprague–Dawley rats

(2010) Kim Rijkers et al.   EPILEPSY RESEARCH

The neurexin superfamily of Caenorhabditis elegans

(2010) Liat Haklai-Topper et al.   GENE EXPRESSION PATTERNS

TUBA1A mutations cause wide spectrum lissencephaly (smooth brain) and suggest that multiple neuronal migration pathways converge on alpha tubulins

(2010) Ravinesh A. Kumar et al.   HUMAN MOLECULAR GENETICS

High-throughput study of synaptic transmission at the neuromuscular junction enabled by optogenetics and microfluidics

(2010) Jeffrey N. Stirman et al.   JOURNAL OF NEUROSCIENCE METHODS

A Functional Link between the Histone Demethylase PHF8 and the Transcription Factor ZNF711 in X-Linked Mental Retardation

(2010) Daniela Kleine-Kohlbrecher et al.   MOLECULAR CELL

ANK repeat-domain of SHN-1 Is indispensable for in vivo SHN-1 function in C. elegans

(2010) Won Chan Oh et al.   MOLECULES AND CELLS

Deletion of the potassium channel Kv12.2 causes hippocampal hyperexcitability and epilepsy

(2010) Xiaofei Zhang et al.   NATURE NEUROSCIENCE

Motor Neuron Synapse and Axon Defects in a C. elegans Alpha-Tubulin Mutant

(2010) Renee Baran et al.   PLoS One

Mutations in mouse Aspm (abnormal spindle-like microcephaly associated) cause not only microcephaly but also major defects in the germline

(2010) J. N. Pulvers et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Baccoside A suppresses epileptic-like seizure/convulsion in Caenorhabditis elegans

(2010) Rakesh Pandey et al.   SEIZURE-EUROPEAN JOURNAL OF EPILEPSY

In the swim of things: recent insights to neurogenetic disorders from zebrafish

(2010) Edor Kabashi et al.   TRENDS IN GENETICS

De novoSTXBP1mutations in mental retardation and nonsyndromic epilepsy

(2009) Fadi F. Hamdan et al.   ANNALS OF NEUROLOGY

Schizophrenia-relevant behaviours in a genetic mouse model for Y2 deficiency

(2009) Tim Karl et al.   BEHAVIOURAL BRAIN RESEARCH

ATX-3, CDC-48 and UBXN-5: A new trimolecular complex in Caenorhabditis elegans

(2009) Ana-João Rodrigues et al.   BIOCHEMICAL AND BIOPHYSICAL RESEARCH COMMUNICATIONS

Function and regulation of Dyrk1A: towards understanding Down syndrome

(2009) Joongkyu Park et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Pharmacogenetic Analysis Reveals a Post-Developmental Role for Rac GTPases in Caenorhabditis elegans GABAergic Neurotransmission

(2009) Cody J. Locke et al.   GENETICS

Familial and sporadic 15q13.3 microdeletions in idiopathic generalized epilepsy: precedent for disorders with complex inheritance

(2009) L. M. Dibbens et al.   HUMAN MOLECULAR GENETICS

SUT-2 potentiates tau-induced neurotoxicity in Caenorhabditis elegans

(2009) Chris R. Guthrie et al.   HUMAN MOLECULAR GENETICS

Abnormal Network Activity in a Targeted Genetic Model of Human Double Cortex

(2009) J. B. Ackman et al.   JOURNAL OF NEUROSCIENCE

Neurobeachin, a protein implicated in membrane protein traffic and autism, is required for the formation and functioning of central synapses

(2009) Lucian Medrihan et al.   JOURNAL OF PHYSIOLOGY-LONDON

A hub-and-spoke circuit drives pheromone attraction and social behaviour in C. elegans

(2009) Evan Z. Macosko et al.   NATURE

NuMA-related LIN-5, ASPM-1, calmodulin and dynein promote meiotic spindle rotation independently of cortical LIN-5/GPR/Gα

(2009) Monique van der Voet et al.   NATURE CELL BIOLOGY

15q13.3 microdeletions increase risk of idiopathic generalized epilepsy

(2009) Ingo Helbig et al.   NATURE GENETICS

Genes associated with idiopathic epilepsies: a current overview

(2009) Yang Lu et al.   NEUROLOGICAL RESEARCH

Nematode Homologue of PQBP1, a Mental Retardation Causative Gene, Is Involved in Lipid Metabolism

(2009) Keiko Takahashi et al.   PLoS One

Green Tea Polyphenols Rescue of Brain Defects Induced by Overexpression of DYRK1A

(2009) Fayçal Guedj et al.   PLoS One

Cocaine Modulates Locomotion Behavior in C. elegans

(2009) Alex Ward et al.   PLoS One

Mice lacking doublecortin and doublecortin-like kinase 2 display altered hippocampal neuronal maturation and spontaneous seizures

(2009) G. Kerjan et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Graded synaptic transmission at the Caenorhabditis elegans neuromuscular junction

(2009) Q. Liu et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Analysis of the neuroligin 4Y gene in patients with autism

(2009) Jin Yan et al.   PSYCHIATRIC GENETICS

Axon Regeneration Requires a Conserved MAP Kinase Pathway

(2009) M. Hammarlund et al.   SCIENCE

Tubulin-related cortical dysgeneses: microtubule dysfunction underlying neuronal migration defects

(2009) Xavier H. Jaglin et al.   TRENDS IN GENETICS

Valproic acid extends Caenorhabditis elegans lifespan

(2008) Kimberley Evason et al.   AGING CELL

Disruption of Neurexin 1 Associated with Autism Spectrum Disorder

(2008) Hyung-Goo Kim et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Targeting Dyrk1A with AAVshRNA Attenuates Motor Alterations in TgDyrk1A, a Mouse Model of Down Syndrome

(2008) Jon Ortiz-Abalia et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Neuronal nicotinic acetylcholine receptors: From the genetic analysis to neurological diseases

(2008) O.K. Steinlein et al.   BIOCHEMICAL PHARMACOLOGY

The immunoglobulin superfamily of neuronal cell adhesion molecules: Lessons from animal models and correlation with human disease

(2008) Markella Katidou et al.   Biotechnology Journal

npr-1 Regulates Foraging and Dispersal Strategies in Caenorhabditis elegans

(2008) Andrea Gloria-Soria et al.   CURRENT BIOLOGY

Mental retardation and associated neurological dysfunctions in Down syndrome: A consequence of dysregulation in critical chromosome 21 genes and associated molecular pathways

(2008) Mohammed Rachidi et al.   EUROPEAN JOURNAL OF PAEDIATRIC NEUROLOGY

Genetic and epigenetic defects in mental retardation

(2008) Jamie M. Kramer et al.   INTERNATIONAL JOURNAL OF BIOCHEMISTRY & CELL BIOLOGY

Association of the neuronal cell adhesion molecule (NRCAM) gene variants with autism

(2008) Tetsuya Marui et al.   INTERNATIONAL JOURNAL OF NEUROPSYCHOPHARMACOLOGY

TheC. elegansL1CAM homologue LAD-2 functions as a coreceptor in MAB-20/Sema2–mediated axon guidance

(2008) Xuelin Wang et al.   JOURNAL OF CELL BIOLOGY

Smaller Dendritic Spines, Weaker Synaptic Transmission, but Enhanced Spatial Learning in Mice Lacking Shank1

(2008) A. Y. Hung et al.   JOURNAL OF NEUROSCIENCE

Trio Mediates Netrin-1-Induced Rac1 Activation in Axon Outgrowth and Guidance

(2008) A. Briancon-Marjollet et al.   MOLECULAR AND CELLULAR BIOLOGY

UNC-18 Promotes Both the Anterograde Trafficking and Synaptic Function of Syntaxin

(2008) Jason M. McEwen et al.   MOLECULAR BIOLOGY OF THE CELL

A variant associated with nicotine dependence, lung cancer and peripheral arterial disease

(2008) Thorgeir E. Thorgeirsson et al.   NATURE

De novo mutations in the gene encoding STXBP1 (MUNC18-1) cause early infantile epileptic encephalopathy

(2008) Hirotomo Saitsu et al.   NATURE GENETICS

Optogenetic analysis of synaptic function

(2008) Jana F Liewald et al.   NATURE METHODS

An RNAi Screen Identifies Genes that Regulate GABA Synapses

(2008) Amy B. Vashlishan et al.   NEURON

Hypothesis-based RNAi screening identifies neuroprotective genes in a Parkinson's disease model

(2008) S. Hamamichi et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Conformational Switch of Syntaxin-1 Controls Synaptic Vesicle Fusion

(2008) Stefan H. Gerber et al.   SCIENCE

Temperature Sensing by an Olfactory Neuron in a Circuit Controlling Behavior of C. elegans

(2008) A. Kuhara et al.   SCIENCE

Intestinal Ca2+ wave dynamics in freely moving C. elegans coordinate execution of a rhythmic motor program

(2007) K. Nehrke et al.   AMERICAN JOURNAL OF PHYSIOLOGY-CELL PHYSIOLOGY

NFAT signaling in neural development and axon growth

(2007) Tuan Nguyen et al.   INTERNATIONAL JOURNAL OF DEVELOPMENTAL NEUROSCIENCE

Functional dissection of the C. elegans cell adhesion molecule SAX-7, a homologue of human L1

(2007) Roger Pocock et al.   MOLECULAR AND CELLULAR NEUROSCIENCE