Small organelle, big responsibility: the role of centrosomes in development and disease

STIL Microcephaly Mutations Interfere with APC/C-Mediated Degradation and Cause Centriole Amplification

(2014) Christian Arquint et al.   CURRENT BIOLOGY

Conserved TCP domain of Sas-4/CPAP is essential for pericentriolar material tethering during centrosome biogenesis

(2014) X. Zheng et al.   PROCEEDINGS OF THE NATIONAL ACADEMY OF SCIENCES OF THE UNITED STATES OF AMERICA

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

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

Dynamics of Hippocampal Neurogenesis in Adult Humans

(2013) Kirsty L. Spalding et al.   CELL

Selective Chemical Crosslinking Reveals a Cep57-Cep63-Cep152 Centrosomal Complex

(2013) Gražvydas Lukinavičius et al.   CURRENT BIOLOGY

Mechanisms of spindle positioning: cortical force generators in the limelight

(2013) Sachin Kotak et al.   CURRENT OPINION IN CELL BIOLOGY

A new cellular stress response that triggers centriolar satellite reorganization and ciliogenesis

(2013) Bine H Villumsen et al.   EMBO JOURNAL

Human microcephaly protein CEP135 binds to hSAS-6 and CPAP, and is required for centriole assembly

(2013) Yu-Chih Lin et al.   EMBO JOURNAL

Centriole distal appendages promote membrane docking, leading to cilia initiation

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

Loss of centrioles causes chromosomal instability in vertebrate somatic cells

(2013) Joo-Hee Sir et al.   JOURNAL OF CELL BIOLOGY

Functional interaction between autophagy and ciliogenesis

(2013) Olatz Pampliega et al.   NATURE

Autophagy promotes primary ciliogenesis by removing OFD1 from centriolar satellites

(2013) Zaiming Tang et al.   NATURE

Cerebral organoids model human brain development and microcephaly

(2013) Madeline A. Lancaster et al.   NATURE

The microcephaly protein Asp regulates neuroepithelium morphogenesis by controlling the spatial distribution of myosin II

(2013) Maria A. Rujano et al.   NATURE CELL BIOLOGY

Centrosome amplification causes microcephaly

(2013) Véronique Marthiens et al.   NATURE CELL BIOLOGY

Maintaining genome stability in the nervous system

(2013) Peter J McKinnon   NATURE NEUROSCIENCE

Structural Analysis of the G-Box Domain of the Microcephaly Protein CPAP Suggests a Role in Centriole Architecture

(2013) Georgios N. Hatzopoulos et al.   STRUCTURE

Microcephaly models in the developing zebrafish retinal neuroepithelium point to an underlying defect in metaphase progression

(2013) C. Novorol et al.   Open Biology

Deficiency in Origin Licensing Proteins Impairs Cilia Formation: Implications for the Aetiology of Meier-Gorlin Syndrome

(2013) Tom Stiff et al.   PLoS Genetics

Crystal structures of the CPAP/STIL complex reveal its role in centriole assembly and human microcephaly

(2013) Matthew A Cottee et al.   eLife

A Truncating Mutation of CEP135 Causes Primary Microcephaly and Disturbed Centrosomal Function

(2012) Muhammad Sajid Hussain et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Exome Capture Reveals ZNF423 and CEP164 Mutations, Linking Renal Ciliopathies to DNA Damage Response Signaling

(2012) Moumita Chaki et al.   CELL

Interkinetic nuclear migration: beyond a hallmark of neurogenesis

(2012) Yoichi Kosodo   CELLULAR AND MOLECULAR LIFE SCIENCES

BLD10/CEP135 Is a Microtubule-Associated Protein that Controls the Formation of the Flagellum Central Microtubule Pair

(2012) Zita Carvalho-Santos et al.   DEVELOPMENTAL CELL

Bardet–Biedl syndrome

(2012) Elizabeth Forsythe et al.   EUROPEAN JOURNAL OF HUMAN GENETICS

Meier-Gorlin syndrome mutations disrupt an Orc1 CDK inhibitory domain and cause centrosome reduplication

(2012) M. Hossain et al.   GENES & DEVELOPMENT

Kinetochore KMN network gene CASC5 mutated in primary microcephaly

(2012) A. Genin et al.   HUMAN MOLECULAR GENETICS

Primary Microcephaly, Impaired DNA Replication, and Genomic Instability Caused by Compound HeterozygousATRMutations

(2012) Houda Mokrani-Benhelli et al.   HUMAN MUTATION

WD40-repeat protein 62 is a JNK-phosphorylated spindle pole protein required for spindle maintenance and timely mitotic progression

(2012) M. A. Bogoyevitch et al.   JOURNAL OF CELL SCIENCE

The centriolar satellite proteins Cep72 and Cep290 interact and are required for recruitment of BBS proteins to the cilium

(2012) Timothy R. Stowe et al.   MOLECULAR BIOLOGY OF THE CELL

Towards a molecular architecture of centriole assembly

(2012) Pierre Gönczy   NATURE REVIEWS MOLECULAR CELL BIOLOGY

Ofd1 Controls Dorso-Ventral Patterning and Axoneme Elongation during Embryonic Brain Development

(2012) Anna D'Angelo et al.   PLoS One

The Centrosome in Cells and Organisms

(2012) M. Bornens   SCIENCE

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

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

Identification of the First ATRIP–Deficient Patient and Novel Mutations in ATR Define a Clinical Spectrum for ATR–ATRIP Seckel Syndrome

(2012) Tomoo Ogi et al.   PLoS Genetics

CPAP is required for cilia formation in neuronal cells

(2012) K.-S. Wu et al.   Biology Open

Whole-exome sequencing identifies compound heterozygous mutations in WDR62 in siblings with recurrent polymicrogyria

(2011) David R. Murdock et al.   AMERICAN JOURNAL OF MEDICAL GENETICS PART A

Primary cilia and organogenesis: is Hedgehog the only sculptor?

(2011) Evangelia Tasouri et al.   CELL AND TISSUE RESEARCH

The human microcephaly protein STIL interacts with CPAP and is required for procentriole formation

(2011) Chieh-Ju C Tang et al.   EMBO JOURNAL

Mechanisms and pathways of growth failure in primordial dwarfism

(2011) A. Klingseisen et al.   GENES & DEVELOPMENT

Strange as it may seem: the many links between Wnt signaling, planar cell polarity, and cilia

(2011) J. B. Wallingford et al.   GENES & DEVELOPMENT

ASPM regulates Wnt signaling pathway activity in the developing brain

(2011) J. J. Buchman et al.   GENES & DEVELOPMENT

The N-terminal region of centrosomal protein 290 (CEP290) restores vision in a zebrafish model of human blindness

(2011) Lisa M. Baye et al.   HUMAN MOLECULAR GENETICS

Nuclear Chk1 prevents premature mitotic entry

(2011) M. Matsuyama et al.   JOURNAL OF CELL SCIENCE

Centriolar satellites are assembly points for proteins implicated in human ciliopathies, including oral-facial-digital syndrome 1

(2011) C. A. M. Lopes et al.   JOURNAL OF CELL SCIENCE

Spindle positioning in human cells relies on proper centriole formation and on the microcephaly proteins CPAP and STIL

(2011) D. Kitagawa et al.   JOURNAL OF CELL SCIENCE

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

Centrosomes, microtubules and neuronal development

(2011) Marijn Kuijpers et al.   MOLECULAR AND CELLULAR NEUROSCIENCE

Timing of centrosome separation is important for accurate chromosome segregation

(2011) William T. Silkworth et al.   MOLECULAR BIOLOGY OF THE CELL

MCPH1 regulates the neuroprogenitor division mode by coupling the centrosomal cycle with mitotic entry through the Chk1–Cdc25 pathway

(2011) Ralph Gruber et al.   NATURE CELL BIOLOGY

Mutations in CEP57 cause mosaic variegated aneuploidy syndrome

(2011) Katie Snape et al.   NATURE GENETICS

A primary microcephaly protein complex forms a ring around parental centrioles

(2011) Joo-Hee Sir et al.   NATURE GENETICS

Mutations in origin recognition complex gene ORC4 cause Meier-Gorlin syndrome

(2011) Duane L Guernsey et al.   NATURE GENETICS

Mutations in ORC1, encoding the largest subunit of the origin recognition complex, cause microcephalic primordial dwarfism resembling Meier-Gorlin syndrome

(2011) Louise S Bicknell et al.   NATURE GENETICS

Defective Wnt-dependent cerebellar midline fusion in a mouse model of Joubert syndrome

(2011) Madeline A Lancaster et al.   NATURE MEDICINE

Ciliogenesis: building the cell's antenna

(2011) Hiroaki Ishikawa et al.   NATURE REVIEWS MOLECULAR CELL BIOLOGY

Autosomal recessive primary microcephaly (MCPH): clinical manifestations, genetic heterogeneity and mutation continuum

(2011) Saqib Mahmood et al.   Orphanet Journal of Rare Diseases

Ciliopathies: an expanding disease spectrum

(2011) Aoife M. Waters et al.   PEDIATRIC NEPHROLOGY

Cdk5rap2 exposes the centrosomal root of microcephaly syndromes

(2011) Timothy L. Megraw et al.   TRENDS IN CELL BIOLOGY

Centrosomes and cilia in human disease

(2011) Mónica Bettencourt-Dias et al.   TRENDS IN GENETICS

The aneuploidy paradox: costs and benefits of an incorrect karyotype

(2011) Jason M. Sheltzer et al.   TRENDS IN GENETICS

Sas-4 provides a scaffold for cytoplasmic complexes and tethers them in a centrosome

(2011) Jayachandran Gopalakrishnan et al.   Nature Communications

Neural stem and progenitor cells shorten S-phase on commitment to neuron production

(2011) Yoko Arai et al.   Nature Communications

CtIP Mutations Cause Seckel and Jawad Syndromes

(2011) Per Qvist et al.   PLoS Genetics

Mutations in Centrosomal Protein CEP152 in Primary Microcephaly Families Linked to MCPH4

(2010) Duane L. Guernsey et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Human ASPM participates in spindle organisation, spindle orientation and cytokinesis

(2010) Julie Higgins et al.   BMC CELL BIOLOGY

Prolonged Prometaphase Blocks Daughter Cell Proliferation Despite Normal Completion of Mitosis

(2010) Yumi Uetake et al.   CURRENT BIOLOGY

Centriole Reduplication during Prolonged Interphase Requires Procentriole Maturation Governed by Plk1

(2010) Jadranka Lončarek et al.   CURRENT BIOLOGY

Centrioles Regulate Centrosome Size by Controlling the Rate of Cnn Incorporation into the PCM

(2010) Paul T. Conduit et al.   CURRENT BIOLOGY

Neural stem cells: the need for a proper orientation

(2010) Bart Lesage et al.   CURRENT OPINION IN GENETICS & DEVELOPMENT

Cdk5rap2 regulates centrosome function and chromosome segregation in neuronal progenitors

(2010) S. B. Lizarraga et al.   DEVELOPMENT

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

(2010) Veena Singla et al.   DEVELOPMENTAL CELL

DSas-6 and Ana2 Coassemble into Tubules to Promote Centriole Duplication and Engagement

(2010) Naomi R. Stevens et al.   DEVELOPMENTAL CELL

CEP290, a gene with many faces: mutation overview and presentation of CEP290base

(2010) Frauke Coppieters et al.   HUMAN MUTATION

CEP290 tethers flagellar transition zone microtubules to the membrane and regulates flagellar protein content

(2010) Branch Craige et al.   JOURNAL OF CELL BIOLOGY

Cep152 acts as a scaffold for recruitment of Plk4 and CPAP to the centrosome

(2010) Onur Cizmecioglu et al.   JOURNAL OF CELL BIOLOGY

CDK5RAP2 functions in centrosome to spindle pole attachment and DNA damage response

(2010) Alexis R. Barr et al.   JOURNAL OF CELL BIOLOGY

Novel CENPJ mutation causes Seckel syndrome

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

Whole-exome sequencing identifies recessive WDR62 mutations in severe brain malformations

(2010) Kaya Bilgüvar et al.   NATURE

Asterless is a scaffold for the onset of centriole assembly

(2010) Nikola S. Dzhindzhev et al.   NATURE

Neurogenic radial glia in the outer subventricular zone of human neocortex

(2010) David V. Hansen et al.   NATURE

CEP152 is a genome maintenance protein disrupted in Seckel syndrome

(2010) Ersan Kalay et al.   NATURE GENETICS

WDR62 is associated with the spindle pole and is mutated in human microcephaly

(2010) Adeline K Nicholas et al.   NATURE GENETICS

OSVZ progenitors of human and ferret neocortex are epithelial-like and expand by integrin signaling

(2010) Simone A Fietz et al.   NATURE NEUROSCIENCE

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

OFD1 Is Mutated in X-Linked Joubert Syndrome and Interacts with LCA5-Encoded Lebercilin

(2009) Karlien L.M. Coene et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Mutations in STIL, Encoding a Pericentriolar and Centrosomal Protein, Cause Primary Microcephaly

(2009) Arun Kumar et al.   AMERICAN JOURNAL OF HUMAN GENETICS

Centriole Age Underlies Asynchronous Primary Cilium Growth in Mammalian Cells

(2009) Charles T. Anderson et al.   CURRENT BIOLOGY

Control of Centriole Length by CPAP and CP110

(2009) Thorsten I. Schmidt et al.   CURRENT BIOLOGY

Overly Long Centrioles and Defective Cell Division upon Excess of the SAS-4-Related Protein CPAP

(2009) Gregor Kohlmaier et al.   CURRENT BIOLOGY

The nonmotile ciliopathies

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

Microcephalin and pericentrin regulate mitotic entry via centrosome-associated Chk1

(2009) Alexandra Tibelius et al.   JOURNAL OF CELL BIOLOGY

A mechanism linking extra centrosomes to chromosomal instability

(2009) Neil J. Ganem et al.   NATURE

CPAP is a cell-cycle regulated protein that controls centriole length

(2009) Chieh-Ju C. Tang et al.   NATURE CELL BIOLOGY

Multipolar Spindle Pole Coalescence Is a Major Source of Kinetochore Mis-Attachment and Chromosome Mis-Segregation in Cancer Cells

(2009) William T. Silkworth et al.   PLoS One

Primary microcephaly: do all roads lead to Rome?

(2009) Gemma K. Thornton et al.   TRENDS IN GENETICS

Regulation of Neurogenesis by Interkinetic Nuclear Migration through an Apical-Basal Notch Gradient

(2008) Filippo Del Bene et al.   CELL

CP110 Suppresses Primary Cilia Formation through Its Interaction with CEP290, a Protein Deficient in Human Ciliary Disease

(2008) William Y. Tsang et al.   DEVELOPMENTAL CELL

Mechanisms to suppress multipolar divisions in cancer cells with extra centrosomes

(2008) M. Kwon et al.   GENES & DEVELOPMENT

Multiple centrosomes: together they stand, divided they fall

(2008) F. Gergely et al.   GENES & DEVELOPMENT

Drosophila asterless and Vertebrate Cep152 Are Orthologs Essential for Centriole Duplication

(2008) S. Blachon et al.   GENETICS

Genetic interaction between Bardet-Biedl syndrome genes and implications for limb patterning

(2008) Marwan K. Tayeh et al.   HUMAN MOLECULAR GENETICS

Convergent extension movements and ciliary function are mediated by ofd1 , a zebrafish orthologue of the human oral-facial-digital type 1 syndrome gene

(2008) Maria I. Ferrante et al.   HUMAN MOLECULAR GENETICS

CEP290 interacts with the centriolar satellite component PCM-1 and is required for Rab8 localization to the primary cilium

(2008) Joon Kim et al.   HUMAN MOLECULAR GENETICS

Extra centrosomes and/or chromosomes prolong mitosis in human cells

(2008) Zhenye Yang et al.   NATURE CELL BIOLOGY

Control of daughter centriole formation by the pericentriolar material

(2008) Jadranka Loncarek et al.   NATURE CELL BIOLOGY

Mutations in the Pericentrin (PCNT) Gene Cause Primordial Dwarfism

(2008) A. Rauch et al.   SCIENCE