Search results for "siRNA screen"

showing 2 items of 2 documents

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

2015

Item does not contain fulltext Defects in primary cilium biogenesis underlie the ciliopathies, a growing group of genetic disorders. We describe a whole-genome siRNA-based reverse genetics screen for defects in biogenesis and/or maintenance of the primary cilium, obtaining a global resource. We identify 112 candidate ciliogenesis and ciliopathy genes, including 44 components of the ubiquitin-proteasome system, 12 G-protein-coupled receptors, and 3 pre-mRNA processing factors (PRPF6, PRPF8 and PRPF31) mutated in autosomal dominant retinitis pigmentosa. The PRPFs localize to the connecting cilium, and PRPF8- and PRPF31-mutated cells have ciliary defects. Combining the screen with exome sequen…

PRPF31Pregnancy ProteinsInbred C57BLCiliopathiesMiceImmunologicCerebellumDatabases GeneticEye AbnormalitiesNon-U.S. Gov'tZebrafishExome sequencingMice KnockoutGeneticsResearch Support Non-U.S. Gov'tCiliumHigh-Throughput Nucleotide SequencingMetabolic Disorders Radboud Institute for Molecular Life Sciences [Radboudumc 6]GenomicsKidney Diseases CysticPhenotypeKidney DiseasesRNA InterferenceAbnormalitiesMultipleFunctional genomicsCiliary Motility DisordersGenetic MarkersEllis-Van Creveld SyndromeKnockoutJeune syndromeOther Research Radboud Institute for Molecular Life Sciences [Radboudumc 0]BiologyResearch SupportTransfectionRetinaArticlewhole-genome siRNA screenJoubert syndromeN.I.H.DatabasesCysticreverse geneticsResearch Support N.I.H. ExtramuralGeneticCerebellar DiseasesJoubert syndromeCiliogenesisSuppressor FactorsJournal ArticleSuppressor Factors ImmunologicmedicineAnimalsHumansAbnormalities MultipleGenetic Predisposition to DiseasePhotoreceptor CellsCiliaGenetic TestingCaenorhabditis elegansExtramuralMembrane ProteinsProteinsReproducibility of ResultsCell Biologymedicine.diseaseMice Inbred C57BLCytoskeletal ProteinsCiliopathyRenal disorders Radboud Institute for Molecular Life Sciences [Radboudumc 11]HEK293 CellsMutationciliopathiesGenome-Wide Association StudyNature Cell Biology
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Epigenetic siRNA and Chemical Screens Identify SETD8 Inhibition as a Therapeutic Strategy for p53 Activation in High-Risk Neuroblastoma

2017

Given the paucity of druggable mutations in high-risk neuroblastoma (NB), we undertook chromatin-focused small interfering RNA and chemical screens to uncover epigenetic regulators critical for the differentiation block in high-risk NB. High-content Opera imaging identified 53 genes whose loss of expression led to a decrease in NB cell proliferation and 16 also induced differentiation. From these, the secondary chemical screen identified SETD8, the H4K20me1 methyltransferase, as a druggable NB target. Functional studies revealed that SETD8 ablation rescued the pro-apoptotic and cell-cycle arrest functions of p53 by decreasing p53K382me1, leading to activation of the p53 canonical pathway. I…

p530301 basic medicineCancer ResearchSmall interfering RNAMethyltransferaseCellular differentiationDruggabilityBiologyArticleEpigenesis GeneticNeuroblastoma03 medical and health sciences0302 clinical medicineNeuroblastomamedicineHumansEpigeneticsRNA Small InterferingGeneCell ProliferationsiRNA screenCell growthQuinazolineCell DifferentiationdifferentiationHistone-Lysine N-Methyltransferasemedicine.diseaseSETD8030104 developmental biologyOncology030220 oncology & carcinogenesisQuinazolinesCancer researchdifferentiation; epigenetics; neuroblastoma; p53; SETD8; siRNA screen; Oncology; Cell Biology; Cancer ResearchTumor Suppressor Protein p53epigeneticHuman
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