Systematic genetic mapping of necroptosis identifies SLC39A7 as modulator of death receptor trafficking.


Journal

Cell death and differentiation
ISSN: 1476-5403
Titre abrégé: Cell Death Differ
Pays: England
ID NLM: 9437445

Informations de publication

Date de publication:
06 2019
Historique:
received: 26 03 2018
accepted: 22 07 2018
revised: 04 07 2018
pubmed: 22 9 2018
medline: 2 10 2020
entrez: 22 9 2018
Statut: ppublish

Résumé

Regulation of cell and tissue homeostasis by programmed cell death is a fundamental process with wide physiological and pathological implications. The advent of scalable somatic cell genetic technologies creates the opportunity to functionally map such essential pathways, thereby identifying potential disease-relevant components. We investigated the genetic basis underlying necroptotic cell death by performing a complementary set of loss-of-function and gain-of-function genetic screens. To this end, we established FADD-deficient haploid human KBM7 cells, which specifically and efficiently undergo necroptosis after a single treatment with either TNFα or the SMAC mimetic compound birinapant. A series of unbiased gene-trap screens identified key signaling mediators, such as TNFR1, RIPK1, RIPK3, and MLKL. Among the novel components, we focused on the zinc transporter SLC39A7, whose knock-out led to necroptosis resistance by affecting TNF receptor surface levels. Orthogonal, solute carrier (SLC)-focused CRISPR/Cas9-based genetic screens revealed the exquisite specificity of SLC39A7, among ~400 SLC genes, for TNFR1-mediated and FAS-mediated but not TRAIL-R1-mediated responses. Mechanistically, we demonstrate that loss of SLC39A7 resulted in augmented ER stress and impaired receptor trafficking, thereby globally affecting downstream signaling. The newly established cellular model also allowed genome-wide gain-of-function screening for genes conferring resistance to necroptosis via the CRISPR/Cas9-based synergistic activation mediator approach. Among these, we found cIAP1 and cIAP2, and characterized the role of TNIP1, which prevented pathway activation in a ubiquitin-binding dependent manner. Altogether, the gain-of-function and loss-of-function screens described here provide a global genetic chart of the molecular factors involved in necroptosis and death receptor signaling, prompting further investigation of their individual contribution and potential role in pathological conditions.

Identifiants

pubmed: 30237509
doi: 10.1038/s41418-018-0192-6
pii: 10.1038/s41418-018-0192-6
pmc: PMC6748104
doi:

Substances chimiques

Cation Transport Proteins 0
Receptors, Tumor Necrosis Factor, Type I 0
SLC39A7 protein, human 0
Tumor Necrosis Factor-alpha 0

Types de publication

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

Langues

eng

Sous-ensembles de citation

IM

Pagination

1138-1155

Subventions

Organisme : Austrian Science Fund FWF
ID : P 29250
Pays : Austria

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Auteurs

Astrid Fauster (A)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria.

Manuele Rebsamen (M)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria. mrebsamen@cemm.oeaw.ac.at.

Katharina L Willmann (KL)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria.
Ludwig Boltzmann Institute for Rare and Undiagnosed Diseases, 1090, Vienna, Austria.

Adrian César-Razquin (A)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria.

Enrico Girardi (E)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria.

Johannes W Bigenzahn (JW)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria.

Fiorella Schischlik (F)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria.

Stefania Scorzoni (S)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria.

Manuela Bruckner (M)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria.

Justyna Konecka (J)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria.

Katrin Hörmann (K)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria.

Leonhard X Heinz (LX)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria.

Kaan Boztug (K)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria.
Ludwig Boltzmann Institute for Rare and Undiagnosed Diseases, 1090, Vienna, Austria.
Department of Pediatrics and Adolescent Medicine, Medical University of Vienna, 1090, Vienna, Austria.
Department of Pediatrics, St. Anna Kinderspital and Children's Cancer Research Institute, Medical University of Vienna, 1090, Vienna, Austria.

Giulio Superti-Furga (G)

CeMM Research Center for Molecular Medicine of the Austrian Academy of Sciences, 1090, Vienna, Austria. gsuperti@cemm.oeaw.ac.at.
Center for Physiology and Pharmacology, Medical University of Vienna, Vienna, Austria. gsuperti@cemm.oeaw.ac.at.

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