Parallel reverse genetic screening in mutant human cells using transcriptomics

Abstract Reverse genetic screens have driven gene annotation and target discovery in model organisms. However, many disease‐relevant genotypes and phenotypes cannot be studied in lower organisms. It is therefore essential to overcome technical hurdles associated with large‐scale reverse genetics in...

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Main Authors: Bianca V Gapp, Tomasz Konopka, Thomas Penz, Vineet Dalal, Tilmann Bürckstümmer, Christoph Bock, Sebastian MB Nijman
Format: Article
Language:English
Published: Springer Nature 2016-08-01
Series:Molecular Systems Biology
Subjects:
Online Access:https://doi.org/10.15252/msb.20166890
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author Bianca V Gapp
Tomasz Konopka
Thomas Penz
Vineet Dalal
Tilmann Bürckstümmer
Christoph Bock
Sebastian MB Nijman
author_facet Bianca V Gapp
Tomasz Konopka
Thomas Penz
Vineet Dalal
Tilmann Bürckstümmer
Christoph Bock
Sebastian MB Nijman
author_sort Bianca V Gapp
collection DOAJ
description Abstract Reverse genetic screens have driven gene annotation and target discovery in model organisms. However, many disease‐relevant genotypes and phenotypes cannot be studied in lower organisms. It is therefore essential to overcome technical hurdles associated with large‐scale reverse genetics in human cells. Here, we establish a reverse genetic approach based on highly robust and sensitive multiplexed RNA sequencing of mutant human cells. We conduct 10 parallel screens using a collection of engineered haploid isogenic cell lines with knockouts covering tyrosine kinases and identify known and unexpected effects on signaling pathways. Our study provides proof of concept for a scalable approach to link genotype to phenotype in human cells, which has broad applications. In particular, it clears the way for systematic phenotyping of still poorly characterized human genes and for systematic study of uncharacterized genomic features associated with human disease.
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institution DOAJ
issn 1744-4292
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publishDate 2016-08-01
publisher Springer Nature
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spelling doaj-art-d8ea184279a4405fb6d0eab253cd88442025-08-20T03:06:27ZengSpringer NatureMolecular Systems Biology1744-42922016-08-011281910.15252/msb.20166890Parallel reverse genetic screening in mutant human cells using transcriptomicsBianca V Gapp0Tomasz Konopka1Thomas Penz2Vineet Dalal3Tilmann Bürckstümmer4Christoph Bock5Sebastian MB Nijman6Nuffield Department of Clinical Medicine, Ludwig Cancer Research Ltd., University of OxfordNuffield Department of Clinical Medicine, Ludwig Cancer Research Ltd., University of OxfordCeMM Research Center for Molecular Medicine of the Austrian Academy of SciencesNuffield Department of Clinical Medicine, Ludwig Cancer Research Ltd., University of OxfordHorizon GenomicsCeMM Research Center for Molecular Medicine of the Austrian Academy of SciencesNuffield Department of Clinical Medicine, Ludwig Cancer Research Ltd., University of OxfordAbstract Reverse genetic screens have driven gene annotation and target discovery in model organisms. However, many disease‐relevant genotypes and phenotypes cannot be studied in lower organisms. It is therefore essential to overcome technical hurdles associated with large‐scale reverse genetics in human cells. Here, we establish a reverse genetic approach based on highly robust and sensitive multiplexed RNA sequencing of mutant human cells. We conduct 10 parallel screens using a collection of engineered haploid isogenic cell lines with knockouts covering tyrosine kinases and identify known and unexpected effects on signaling pathways. Our study provides proof of concept for a scalable approach to link genotype to phenotype in human cells, which has broad applications. In particular, it clears the way for systematic phenotyping of still poorly characterized human genes and for systematic study of uncharacterized genomic features associated with human disease.https://doi.org/10.15252/msb.20166890kinasesmultiplexed RNA sequencingparallel screeningreverse geneticssystematic phenotyping
spellingShingle Bianca V Gapp
Tomasz Konopka
Thomas Penz
Vineet Dalal
Tilmann Bürckstümmer
Christoph Bock
Sebastian MB Nijman
Parallel reverse genetic screening in mutant human cells using transcriptomics
Molecular Systems Biology
kinases
multiplexed RNA sequencing
parallel screening
reverse genetics
systematic phenotyping
title Parallel reverse genetic screening in mutant human cells using transcriptomics
title_full Parallel reverse genetic screening in mutant human cells using transcriptomics
title_fullStr Parallel reverse genetic screening in mutant human cells using transcriptomics
title_full_unstemmed Parallel reverse genetic screening in mutant human cells using transcriptomics
title_short Parallel reverse genetic screening in mutant human cells using transcriptomics
title_sort parallel reverse genetic screening in mutant human cells using transcriptomics
topic kinases
multiplexed RNA sequencing
parallel screening
reverse genetics
systematic phenotyping
url https://doi.org/10.15252/msb.20166890
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AT tomaszkonopka parallelreversegeneticscreeninginmutanthumancellsusingtranscriptomics
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AT vineetdalal parallelreversegeneticscreeninginmutanthumancellsusingtranscriptomics
AT tilmannburckstummer parallelreversegeneticscreeninginmutanthumancellsusingtranscriptomics
AT christophbock parallelreversegeneticscreeninginmutanthumancellsusingtranscriptomics
AT sebastianmbnijman parallelreversegeneticscreeninginmutanthumancellsusingtranscriptomics