A human tissue screen identifies a regulator of ER secretion as a brain-size determinant.


Journal

Science (New York, N.Y.)
ISSN: 1095-9203
Titre abrégé: Science
Pays: United States
ID NLM: 0404511

Informations de publication

Date de publication:
20 11 2020
Historique:
received: 04 03 2020
accepted: 10 09 2020
pubmed: 31 10 2020
medline: 22 1 2021
entrez: 30 10 2020
Statut: ppublish

Résumé

Loss-of-function (LOF) screens provide a powerful approach to identify regulators in biological processes. Pioneered in laboratory animals, LOF screens of human genes are currently restricted to two-dimensional cell cultures, which hinders the testing of gene functions requiring tissue context. Here, we present CRISPR-lineage tracing at cellular resolution in heterogeneous tissue (CRISPR-LICHT), which enables parallel LOF studies in human cerebral organoid tissue. We used CRISPR-LICHT to test 173 microcephaly candidate genes, revealing 25 to be involved in known and uncharacterized microcephaly-associated pathways. We characterized

Identifiants

pubmed: 33122427
pii: science.abb5390
doi: 10.1126/science.abb5390
doi:

Substances chimiques

Carrier Proteins 0
Extracellular Matrix Proteins 0
IER3IP1 protein, human 0
Membrane Proteins 0

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

935-941

Commentaires et corrections

Type : CommentIn

Informations de copyright

Copyright © 2020 The Authors, some rights reserved; exclusive licensee American Association for the Advancement of Science. No claim to original U.S. Government Works.

Auteurs

Christopher Esk (C)

Institute of Molecular Biotechnology of the Austrian Academy of Science (IMBA), Vienna BioCenter (VBC), Vienna, Austria.

Dominik Lindenhofer (D)

Institute of Molecular Biotechnology of the Austrian Academy of Science (IMBA), Vienna BioCenter (VBC), Vienna, Austria.

Simon Haendeler (S)

Institute of Molecular Biotechnology of the Austrian Academy of Science (IMBA), Vienna BioCenter (VBC), Vienna, Austria.
Center for Integrative Bioinformatics Vienna (CIBIV), Max F. Perutz Laboratories, University of Vienna and Medical University of Vienna, VBC, Vienna, Austria.

Roelof A Wester (RA)

Institute of Molecular Biotechnology of the Austrian Academy of Science (IMBA), Vienna BioCenter (VBC), Vienna, Austria.

Florian Pflug (F)

Center for Integrative Bioinformatics Vienna (CIBIV), Max F. Perutz Laboratories, University of Vienna and Medical University of Vienna, VBC, Vienna, Austria.

Benoit Schroeder (B)

Center for Integrative Bioinformatics Vienna (CIBIV), Max F. Perutz Laboratories, University of Vienna and Medical University of Vienna, VBC, Vienna, Austria.

Joshua A Bagley (JA)

Institute of Molecular Biotechnology of the Austrian Academy of Science (IMBA), Vienna BioCenter (VBC), Vienna, Austria.

Ulrich Elling (U)

Institute of Molecular Biotechnology of the Austrian Academy of Science (IMBA), Vienna BioCenter (VBC), Vienna, Austria.

Johannes Zuber (J)

Research Institute of Molecular Pathology (IMP), VBC, Vienna, Austria.
Medical University of Vienna, VBC, Vienna, Austria.

Arndt von Haeseler (A)

Center for Integrative Bioinformatics Vienna (CIBIV), Max F. Perutz Laboratories, University of Vienna and Medical University of Vienna, VBC, Vienna, Austria.
Bioinformatics and Computational Biology, Faculty of Computer Science, University of Vienna, Vienna, Austria.

Jürgen A Knoblich (JA)

Institute of Molecular Biotechnology of the Austrian Academy of Science (IMBA), Vienna BioCenter (VBC), Vienna, Austria. juergen.knoblich@imba.oeaw.ac.at.
Medical University of Vienna, VBC, Vienna, Austria.

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Classifications MeSH