A CRISPR-based screen for Hedgehog signaling provides insights into ciliary function and ciliopathies

RABL2 interacts with the intraflagellar transport-B complex and CEP19 and participates in ciliary assembly

(2017) Yuya Nishijima et al.   MOLECULAR BIOLOGY OF THE CELL

Architecture of the human interactome defines protein communities and disease networks

(2017) Edward L. Huttlin et al.   NATURE

Genome-scale measurement of off-target activity using Cas9 toxicity in high-throughput screens

(2017) David W. Morgens et al.   Nature Communications

CEP19 cooperates with FOP and CEP350 to drive early steps in the ciliogenesis programme

(2017) Bahareh A. Mojarad et al.   Open Biology

A Genome-wide CRISPR Death Screen Identifies Genes Essential for Oxidative Phosphorylation

(2016) Jason D. Arroyo et al.   Cell Metabolism

Hedgehog Signal Transduction: Key Players, Oncogenic Drivers, and Cancer Therapy

(2016) Ekaterina Pak et al.   DEVELOPMENTAL CELL

Centriolar CPAP/SAS-4 Imparts Slow Processive Microtubule Growth

(2016) Ashwani Sharma et al.   DEVELOPMENTAL CELL

BAR Domain-Containing FAM92 Proteins Interact with Chibby1 To Facilitate Ciliogenesis

(2016) Feng-Qian Li et al.   MOLECULAR AND CELLULAR BIOLOGY

Systematic comparison of CRISPR/Cas9 and RNAi screens for essential genes

(2016) David W Morgens et al.   NATURE BIOTECHNOLOGY

Parallel shRNA and CRISPR-Cas9 screens enable antiviral drug target identification

(2016) Richard M Deans et al.   Nature Chemical Biology

Distinct genetic architectures for syndromic and nonsyndromic congenital heart defects identified by exome sequencing

(2016) Alejandro Sifrim et al.   NATURE GENETICS

A genome-wide CRISPR screen identifies a restricted set of HIV host dependency factors

(2016) Ryan J Park et al.   NATURE GENETICS

Discovery of a proteinaceous cellular receptor for a norovirus

(2016) R. C. Orchard et al.   SCIENCE

A mechanism for the elimination of the female gamete centrosome in Drosophila melanogaster

(2016) A. Pimenta-Marques et al.   SCIENCE

Primary Cilia and Mammalian Hedgehog Signaling

(2016) Fiona Bangs et al.   Cold Spring Harbor Perspectives in Biology

Ciliopathies

(2016) Daniela A. Braun et al.   Cold Spring Harbor Perspectives in Biology

Genomic Copy Number Dictates a Gene-Independent Cell Response to CRISPR/Cas9 Targeting

(2016) A. J. Aguirre et al.   Cancer Discovery

A CRISPR Dropout Screen Identifies Genetic Vulnerabilities and Therapeutic Targets in Acute Myeloid Leukemia

(2016) Konstantinos Tzelepis et al.   Cell Reports

A Dynamic Protein Interaction Landscape of the Human Centrosome-Cilium Interface

(2015) Gagan D. Gupta et al.   CELL

High-Resolution CRISPR Screens Reveal Fitness Genes and Genotype-Specific Cancer Liabilities

(2015) Traver Hart et al.   CELL

A Human Interactome in Three Quantitative Dimensions Organized by Stoichiometries and Abundances

(2015) Marco Y. Hein et al.   CELL

Zeta-Tubulin Is a Member of a Conserved Tubulin Module and Is a Component of the Centriolar Basal Foot in Multiciliated Cells

(2015) Erin Turk et al.   CURRENT BIOLOGY

Identification and functional characterization ofde novo FOXP1variants provides novel insights into the etiology of neurodevelopmental disorder

(2015) Elliot Sollis et al.   HUMAN MOLECULAR GENETICS

Matching Two Independent Cohorts ValidatesDPH1as a Gene Responsible for Autosomal Recessive Intellectual Disability with Short Stature, Craniofacial, and Ectodermal Anomalies

(2015) Catrina M. Loucks et al.   HUMAN MUTATION

Global genetic analysis in mice unveils central role for cilia in congenital heart disease

(2015) You Li et al.   NATURE

An siRNA-based functional genomics screen for the identification of regulators of ciliogenesis and ciliopathy genes

(2015) Gabrielle Wheway et al.   NATURE CELL BIOLOGY

The EMBL-EBI bioinformatics web and programmatic tools framework

(2015) Weizhong Li et al.   NUCLEIC ACIDS RESEARCH

Reversible centriole depletion with an inhibitor of Polo-like kinase 4

(2015) Y. L. Wong et al.   SCIENCE

Identification and characterization of essential genes in the human genome

(2015) T. Wang et al.   SCIENCE

Functional genome-wide siRNA screen identifies KIAA0586 as mutated in Joubert syndrome

(2015) Susanne Roosing et al.   eLife

The Intraflagellar Transport Protein IFT27 Promotes BBSome Exit from Cilia through the GTPase ARL6/BBS3

(2014) Gerald M. Liew et al.   DEVELOPMENTAL CELL

EFCAB7 and IQCE Regulate Hedgehog Signaling by Tethering the EVC-EVC2 Complex to the Base of Primary Cilia

(2014) Ganesh V. Pusapati et al.   DEVELOPMENTAL CELL

Chibby promotes ciliary vesicle formation and basal body docking during airway cell differentiation

(2014) Michael C. Burke et al.   JOURNAL OF CELL BIOLOGY

Cby1 promotes Ahi1 recruitment to a ring-shaped domain at the centriole–cilium interface and facilitates proper cilium formation and function

(2014) Yin Loon Lee et al.   MOLECULAR BIOLOGY OF THE CELL

Measuring error rates in genomic perturbation screens: gold standards for human functional genomics

(2014) T. Hart et al.   Molecular Systems Biology

High-throughput screening of a CRISPR/Cas9 library for functional genomics in human cells

(2014) Yuexin Zhou et al.   NATURE

Guidelines for investigating causality of sequence variants in human disease

(2014) D. G. MacArthur et al.   NATURE

Easy quantitative assessment of genome editing by sequence trace decomposition

(2014) Eva K. Brinkman et al.   NUCLEIC ACIDS RESEARCH

How do cilia organize signalling cascades?

(2014) M. V. Nachury   PHILOSOPHICAL TRANSACTIONS OF THE ROYAL SOCIETY B-BIOLOGICAL SCIENCES

Stabilization of Cartwheel-less Centrioles for Duplication Requires CEP295-Mediated Centriole-to-Centrosome Conversion

(2014) Denisse Izquierdo et al.   Cell Reports

Mutations in CSPP1 Lead to Classical Joubert Syndrome

(2013) Naiara Akizu et al.   AMERICAN JOURNAL OF HUMAN GENETICS

CEP120 and SPICE1 Cooperate with CPAP in Centriole Elongation

(2013) David Comartin et al.   CURRENT BIOLOGY

CEP120 interacts with CPAP and positively regulates centriole elongation

(2013) Yi-Nan Lin et al.   JOURNAL OF CELL BIOLOGY

An in vitro assay for entry into cilia reveals unique properties of the soluble diffusion barrier

(2013) David K. Breslow et al.   JOURNAL OF CELL BIOLOGY

 -tubulin is essential in Tetrahymena thermophila for the assembly and stability of basal bodies

(2013) I. Ross et al.   JOURNAL OF CELL SCIENCE

Genome-wide recessive genetic screening in mammalian cells with a lentiviral CRISPR-guide RNA library

(2013) Hiroko Koike-Yusa et al.   NATURE BIOTECHNOLOGY

Genome-Scale CRISPR-Cas9 Knockout Screening in Human Cells

(2013) Ophir Shalem et al.   SCIENCE

Genetic Screens in Human Cells Using the CRISPR-Cas9 System

(2013) Tim Wang et al.   SCIENCE

A Smoothened-Evc2 Complex Transduces the Hedgehog Signal at Primary Cilia

(2012) Karolin V. Dorn et al.   DEVELOPMENTAL CELL

Genomic analysis of Meckel–Gruber syndrome in Arabs reveals marked genetic heterogeneity and novel candidate genes

(2012) Ranad Shaheen et al.   EUROPEAN JOURNAL OF HUMAN GENETICS

Genetic causes of microcephaly and lessons for neuronal development

(2012) Edward C. Gilmore et al.   Wiley Interdisciplinary Reviews-Developmental Biology

Boc and Gas1 Each Form Distinct Shh Receptor Complexes with Ptch1 and Are Required for Shh-Mediated Cell Proliferation

(2011) Luisa Izzi et al.   DEVELOPMENTAL CELL

Overlapping Roles and Collective Requirement for the Coreceptors GAS1, CDO, and BOC in SHH Pathway Function

(2011) Benjamin L. Allen et al.   DEVELOPMENTAL CELL

Novel asymmetrically localizing components of human centrosomes identified by complementary proteomics methods

(2011) Lis Jakobsen et al.   EMBO JOURNAL

Genome-Wide RNAi Screen Reveals Disease-Associated Genes That Are Common to Hedgehog and Wnt Signaling

(2011) L. S. Jacob et al.   Science Signaling

Cep120 is asymmetrically localized to the daughter centriole and is essential for centriole assembly

(2010) Moe R. Mahjoub et al.   JOURNAL OF CELL BIOLOGY

Centriolar Association of ALMS1 and Likely Centrosomal Functions of the ALMS Motif–containing Proteins C10orf90 and KIAA1731

(2010) Victoria J. Knorz et al.   MOLECULAR BIOLOGY OF THE CELL

Functional genomic screen for modulators of ciliogenesis and cilium length

(2010) Joon Kim et al.   NATURE

The primary cilium: a signalling centre during vertebrate development

(2010) Sarah C. Goetz et al.   NATURE REVIEWS GENETICS

Ellis-van Creveld syndrome and Weyers acrodental dysostosis are caused by cilia-mediated diminished response to hedgehog ligands

(2009) Victor L. Ruiz-Perez et al.   AMERICAN JOURNAL OF MEDICAL GENETICS PART C-SEMINARS IN MEDICAL GENETICS

Control of Centriole Length by CPAP and CP110

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

The Zn Finger protein Iguana impacts Hedgehog signaling by promoting ciliogenesis

(2009) Andrew M. Glazer et al.   DEVELOPMENTAL BIOLOGY

Systematic and integrative analysis of large gene lists using DAVID bioinformatics resources

(2009) Da Wei Huang et al.   Nature Protocols

Diphthamide modification of eEF2 requires a J-domain protein and is essential for normal development

(2008) T. R. Webb et al.   JOURNAL OF CELL SCIENCE