The Multifaceted Roles of Primary Cilia in the Development of the Cerebral Cortex

TTBK2 and primary cilia are essential for the connectivity and survival of cerebellar Purkinje neurons

(2020) Emily Bowie et al.   eLife

Loss of CPAP in developing mouse brain and its functional implication in human primary microcephaly

(2020) Yi-Nan Lin et al.   JOURNAL OF CELL SCIENCE

Centrosome anchoring regulates progenitor properties and cortical formation

(2020) Wei Shao et al.   NATURE

Cortical Neural Stem Cell Lineage Progression Is Regulated by Extrinsic Signaling Molecule Sonic Hedgehog

(2020) Yue Zhang et al.   Cell Reports

A discrete subtype of neural progenitor crucial for cortical folding in the gyrencephalic mammalian brain

(2020) Naoyuki Matsumoto et al.   eLife

Developmental and regenerative paradigms of cilia regulated hedgehog signaling

(2020) Daniel Kopinke et al.   SEMINARS IN CELL & DEVELOPMENTAL BIOLOGY

A transient role of the ciliary gene Inpp5e in controlling direct versus indirect neurogenesis in cortical development

(2020) Kerstin Hasenpusch-Theil et al.   eLife

mTOR signaling regulates the morphology and migration of outer radial glia in developing human cortex

(2020) Madeline G Andrews et al.   eLife

Primary cilium-dependent cAMP/PKA signaling at the centrosome regulates neuronal migration

(2020) Julie Stoufflet et al.   Science Advances

Cenpj regulates cilia disassembly and neurogenesis in the developing mouse cortex

(2019) Wenyu Ding et al.   JOURNAL OF NEUROSCIENCE

Derepression of sonic hedgehog signaling upon Gpr161 deletion unravels forebrain and ventricular abnormalities

(2019) Issei S. Shimada et al.   DEVELOPMENTAL BIOLOGY

Roles of Primary Cilia in the Developing Brain

(2019) Sang Min Park et al.   Frontiers in Cellular Neuroscience

Phosphorylation of Ci/Gli by Fused Family Kinases Promotes Hedgehog Signaling

(2019) Yuhong Han et al.   DEVELOPMENTAL CELL

A multiscale mathematical model of cell dynamics during neurogenesis in the mouse cerebral cortex

(2019) Marie Postel et al.   BMC BIOINFORMATICS

Mutations in the Heterotopia Gene Eml1/EML1 Severely Disrupt the Formation of Primary Cilia

(2019) Ana Uzquiano et al.   Cell Reports

Dynamic Changes in Ultrastructure of the Primary Cilium in Migrating Neuroblasts in the Postnatal Brain

(2019) Mami Matsumoto et al.   JOURNAL OF NEUROSCIENCE

Roles for IFT172 and Primary Cilia in Cell Migration, Cell Division, and Neocortex Development

(2019) Michal Pruski et al.   Frontiers in Cell and Developmental Biology

Primary Cilia Signaling Promotes Axonal Tract Development and Is Disrupted in Joubert Syndrome-Related Disorders Models

(2019) Jiami Guo et al.   DEVELOPMENTAL CELL

The Hippo Pathway, YAP/TAZ, and the Plasma Membrane

(2019) Valentina Rausch et al.   TRENDS IN CELL BIOLOGY

Evolution of Cortical Neurogenesis in Amniotes Controlled by Robo Signaling Levels

(2018) Adrián Cárdenas et al.   CELL

Gli2 Rescues Delays in Brain Development Induced by Kif3a Dysfunction

(2018) Jia-Long Chen et al.   CEREBRAL CORTEX

Cell type‐specific regulation of ciliary transition zone assembly in vertebrates

(2018) Antonia Wiegering et al.   EMBO JOURNAL

Brain Somatic Mutations in MTOR Disrupt Neuronal Ciliogenesis, Leading to Focal Cortical Dyslamination

(2018) Sang Min Park et al.   NEURON

Gli3 controls the onset of cortical neurogenesis by regulating the radial glial cell cycle throughCdk6expression

(2018) Kerstin Hasenpusch-Theil et al.   DEVELOPMENT

Altered GLI3 and FGF8 signaling underlies Acrocallosal syndrome phenotypes in Kif7 depleted mice

(2018) Audrey Putoux et al.   HUMAN MOLECULAR GENETICS

Reversal of proliferation deficits caused by chromosome 16p13.11 microduplication through targeting NFκB signaling: an integrated study of patient-derived neuronal precursor cells, cerebral organoids and in vivo brain imaging

(2018) Mandy Johnstone et al.   MOLECULAR PSYCHIATRY

Mutation of the α-tubulin Tuba1a leads to straighter microtubules and perturbs neuronal migration

(2017) Richard Belvindrah et al.   JOURNAL OF CELL BIOLOGY

Super-resolution microscopy reveals that disruption of ciliary transition-zone architecture causes Joubert syndrome

(2017) Xiaoyu Shi et al.   NATURE CELL BIOLOGY

Genes and molecular pathways underpinning ciliopathies

(2017) Jeremy F. Reiter et al.   NATURE REVIEWS MOLECULAR CELL BIOLOGY

Intraflagellar Transport and Ciliary Dynamics

(2017) Hiroaki Ishikawa et al.   Cold Spring Harbor Perspectives in Biology

The intraflagellar transport machinery in ciliary signaling

(2016) André Mourão et al.   CURRENT OPINION IN STRUCTURAL BIOLOGY

mTORC1 signaling and primary cilia are required for brain ventricle morphogenesis

(2016) Philippe Foerster et al.   DEVELOPMENT

CPAP promotes timely cilium disassembly to maintain neural progenitor pool

(2016) Elke Gabriel et al.   EMBO JOURNAL

Hedgehog signaling promotes basal progenitor expansion and the growth and folding of the neocortex

(2016) Lei Wang et al.   NATURE NEUROSCIENCE

Type 3 Adenylyl Cyclase and Somatostatin Receptor 3 Expression Persists in Aged Rat Neocortical and Hippocampal Neuronal Cilia

(2016) Sarah M. Guadiana et al.   Frontiers in Aging Neuroscience

The ciliogenic transcription factor Rfx3 is required for the formation of the thalamocortical tract by regulating the patterning of prethalamus and ventral telencephalon

(2015) Dario Magnani et al.   HUMAN MOLECULAR GENETICS

The role of primary cilia in corpus callosum formation is mediated by production of the Gli3 repressor

(2015) Christine Laclef et al.   HUMAN MOLECULAR GENETICS

The transition zone protein Rpgrip1l regulates proteasomal activity at the primary cilium

(2015) Christoph Gerhardt et al.   JOURNAL OF CELL BIOLOGY

Developmental disruptions underlying brain abnormalities in ciliopathies

(2015) Jiami Guo et al.   Nature Communications

An optogenetic system for interrogating the temporal dynamics of Akt

(2015) Yoshihiro Katsura et al.   Scientific Reports

Suppressor of Fused Is Critical for Maintenance of Neuronal Progenitor Identity during Corticogenesis

(2015) Odessa R. Yabut et al.   Cell Reports

Wnt Signaling Regulates Multipolar-to-Bipolar Transition of Migrating Neurons in the Cerebral Cortex

(2015) Michael Boitard et al.   Cell Reports

Ntf3 acts downstream of Sip1 in cortical postmitotic neurons to control progenitor cell fate through feedback signaling

(2014) S. Parthasarathy et al.   DEVELOPMENT

The kinesin-4 protein Kif7 regulates mammalian Hedgehog signalling by organizing the cilium tip compartment

(2014) Mu He et al.   NATURE CELL BIOLOGY

Thinking out of the dish: what to learn about cortical development using pluripotent stem cells

(2014) Jelle van den Ameele et al.   TRENDS IN NEUROSCIENCES

Gli3 Repressor Controls Cell Fates and Cell Adhesion for Proper Establishment of Neurogenic Niche

(2014) Hui Wang et al.   Cell Reports

Asymmetric Inheritance of Centrosome-Associated Primary Cilium Membrane Directs Ciliogenesis after Cell Division

(2013) Judith T.M.L. Paridaen et al.   CELL

The Ciliary G-Protein-Coupled Receptor Gpr161 Negatively Regulates the Sonic Hedgehog Pathway via cAMP Signaling

(2013) Saikat Mukhopadhyay et al.   CELL

Gli3 is required in Emx1+ progenitors for the development of the corpus callosum

(2013) Eleni-Maria Amaniti et al.   DEVELOPMENTAL BIOLOGY

Centriole distal appendages promote membrane docking, leading to cilia initiation

(2013) B. E. Tanos et al.   GENES & DEVELOPMENT

Titration of GLI3 Repressor Activity by Sonic Hedgehog Signaling Is Critical for Maintaining Multiple Adult Neural Stem Cell and Astrocyte Functions

(2013) R. Petrova et al.   JOURNAL OF NEUROSCIENCE

Filamin A Regulates Neuronal Migration through Brefeldin A-Inhibited Guanine Exchange Factor 2-Dependent Arf1 Activation

(2013) J. Zhang et al.   JOURNAL OF NEUROSCIENCE

Arborization of Dendrites by Developing Neocortical Neurons Is Dependent on Primary Cilia and Type 3 Adenylyl Cyclase

(2013) S. M. Guadiana et al.   JOURNAL OF NEUROSCIENCE

Autophagy promotes primary ciliogenesis by removing OFD1 from centriolar satellites

(2013) Zaiming Tang et al.   NATURE

Primary cilia in neurodevelopmental disorders

(2013) Enza Maria Valente et al.   Nature Reviews Neurology

Branch management: mechanisms of axon branching in the developing vertebrate CNS

(2013) Katherine Kalil et al.   NATURE REVIEWS NEUROSCIENCE

Focal malformations of cortical development: New vistas for molecular pathogenesis

(2013) K.-C. Lim et al.   NEUROSCIENCE

CCDC41 is required for ciliary vesicle docking to the mother centriole

(2013) K. Joo et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Gli3 Controls Corpus Callosum Formation by Positioning Midline Guideposts During Telencephalic Patterning

(2012) Dario Magnani et al.   CEREBRAL CORTEX

Arl13b in Primary Cilia Regulates the Migration and Placement of Interneurons in the Developing Cerebral Cortex

(2012) Holden Higginbotham et al.   DEVELOPMENTAL CELL

The base of the cilium: roles for transition fibres and the transition zone in ciliary formation, maintenance and compartmentalization

(2012) Jeremy F Reiter et al.   EMBO REPORTS

Expanded mutational spectrum of the GLI3 gene substantiates genotype–phenotype correlations

(2012) Aleksander Jamsheer et al.   JOURNAL OF APPLIED GENETICS

Dishevelled stabilization by the ciliopathy protein Rpgrip1l is essential for planar cell polarity

(2012) Alexia Mahuzier et al.   JOURNAL OF CELL BIOLOGY

Migrating fibroblasts reorient directionality by a metastable, PI3K-dependent mechanism

(2012) Erik S. Welf et al.   JOURNAL OF CELL BIOLOGY

Genotype–phenotype correlation inCC2D2A-related Joubert syndrome reveals an association with ventriculomegaly and seizures

(2012) Ruxandra Bachmann-Gagescu et al.   JOURNAL OF MEDICAL GENETICS

Interneuron dysfunction in psychiatric disorders

(2012) Oscar Marín   NATURE REVIEWS NEUROSCIENCE

Tangentially Migrating Neurons Assemble a Primary Cilium that Promotes Their Reorientation to the Cortical Plate

(2012) Jean-Pierre Baudoin et al.   NEURON

Heteromerization of Ciliary G Protein-Coupled Receptors in the Mouse Brain

(2012) Jill A. Green et al.   PLoS One

A Simple Cell-Based Assay Reveals That Diverse Neuropsychiatric Risk Genes Converge on Primary Cilia

(2012) Aaron Marley et al.   PLoS One

Disruption of Mouse Cenpj, a Regulator of Centriole Biogenesis, Phenocopies Seckel Syndrome

(2012) Rebecca E. McIntyre et al.   PLoS Genetics

The Ciliogenic Transcription Factor RFX3 Regulates Early Midline Distribution of Guidepost Neurons Required for Corpus Callosum Development

(2012) Carine Benadiba et al.   PLoS Genetics

Joubert Syndrome and Related Disorders: Spectrum of Neuroimaging Findings in 75 Patients

(2011) A. Poretti et al.   AMERICAN JOURNAL OF NEURORADIOLOGY

Ultrastructure of cilia and flagella - back to the future!

(2011) Cathy Fisch et al.   BIOLOGY OF THE CELL

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

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

Primary cilia control telencephalic patterning and morphogenesis via Gli3 proteolytic processing

(2011) L. Besse et al.   DEVELOPMENT

Basolateral rather than apical primary cilia on neuroepithelial cells committed to delamination

(2011) M. Wilsch-Brauninger et al.   DEVELOPMENT

Primary cilia and Gli3 activity regulate cerebral cortical size

(2011) Sandra L. Wilson et al.   Developmental Neurobiology

A meckelin–filamin A interaction mediates ciliogenesis

(2011) Matthew Adams et al.   HUMAN MOLECULAR GENETICS

Mutations in KIF7 link Joubert syndrome with Sonic Hedgehog signaling and microtubule dynamics

(2011) Claudia Dafinger et al.   JOURNAL OF CLINICAL INVESTIGATION

Oblique Radial Glial Divisions in the Developing Mouse Neocortex Induce Self-Renewing Progenitors outside the Germinal Zone That Resemble Primate Outer Subventricular Zone Progenitors

(2011) A. Shitamukai et al.   JOURNAL OF NEUROSCIENCE

Nde1-mediated inhibition of ciliogenesis affects cell cycle re-entry

(2011) Sehyun Kim et al.   NATURE CELL BIOLOGY

KIF7 mutations cause fetal hydrolethalus and acrocallosal syndromes

(2011) Audrey Putoux et al.   NATURE GENETICS

A transition zone complex regulates mammalian ciliogenesis and ciliary membrane composition

(2011) Francesc R Garcia-Gonzalo et al.   NATURE GENETICS

A new subtype of progenitor cell in the mouse embryonic neocortex

(2011) Xiaoqun Wang et al.   NATURE NEUROSCIENCE

Cilia in the CNS: The Quiet Organelle Claims Center Stage

(2011) Angeliki Louvi et al.   NEURON

Reelin Regulates Cadherin Function via Dab1/Rap1 to Control Neuronal Migration and Lamination in the Neocortex

(2011) Santos J. Franco et al.   NEURON

The neuropathology of autism: defects of neurogenesis and neuronal migration, and dysplastic changes

(2010) Jerzy Wegiel et al.   ACTA NEUROPATHOLOGICA

Transcriptional control of genes involved in ciliogenesis: a first step in making cilia

(2010) Joëlle Thomas et al.   BIOLOGY OF THE CELL

Dopamine receptor 1 localizes to neuronal cilia in a dynamic process that requires the Bardet-Biedl syndrome proteins

(2010) Jacqueline S. Domire et al.   CELLULAR AND MOLECULAR LIFE SCIENCES

Developmental regulation of axon branching in the vertebrate nervous system

(2010) D. A. Gibson et al.   DEVELOPMENT

Ofd1, a Human Disease Gene, Regulates the Length and Distal Structure of Centrioles

(2010) Veena Singla et al.   DEVELOPMENTAL CELL

Novel CENPJ mutation causes Seckel syndrome

(2010) M. S. Al-Dosari et al.   JOURNAL OF MEDICAL GENETICS

Distinct Roles of GABAergic Interneurons in the Regulation of Striatal Output Pathways

(2010) A. H. Gittis et al.   JOURNAL OF NEUROSCIENCE

Kinetics of Hedgehog-Dependent Full-Length Gli3 Accumulation in Primary Cilia and Subsequent Degradation

(2010) X. Wen et al.   MOLECULAR AND CELLULAR BIOLOGY

A synaptic trek to autism

(2009) Thomas Bourgeron   CURRENT OPINION IN NEUROBIOLOGY

The nonmotile ciliopathies

(2009) Jonathan L Tobin et al.   GENETICS IN MEDICINE

INPP5E mutations cause primary cilium signaling defects, ciliary instability and ciliopathies in human and mouse

(2009) Monique Jacoby et al.   NATURE GENETICS

Mutations in INPP5E, encoding inositol polyphosphate-5-phosphatase E, link phosphatidyl inositol signaling to the ciliopathies

(2009) Stephanie L Bielas et al.   NATURE GENETICS

Sip1 regulates sequential fate decisions by feedback signaling from postmitotic neurons to progenitors

(2009) Eve Seuntjens et al.   NATURE NEUROSCIENCE

Mouse Kif7/Costal2 is a cilia-associated protein that regulates Sonic hedgehog signaling

(2009) K. F. Liem et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

Malformations of Cerebral Cortical Development in Oral-Facial-Digital Syndrome Type VI

(2008) J. Takanashi et al.   AMERICAN JOURNAL OF NEURORADIOLOGY

Robust Neuronal Symmetry Breaking by Ras-Triggered Local Positive Feedback

(2008) Marc Fivaz et al.   CURRENT BIOLOGY

Bardet-Biedl syndrome proteins are required for the localization of G protein-coupled receptors to primary cilia

(2008) N. F. Berbari et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA