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@ARTICLE{Meyer:284417,
author = {L. M. Meyer and S. E. Koschade$^*$ and J. B. Vischedyk and
M. Thoelken and A. Gubas and M. Wegner and M. Basoglu and S.
Knapp and M. Kaulich and S. Eimer and S. Shaid$^*$ and C. H.
Brandts$^*$},
title = {{D}eciphering the mitophagy receptor network identifies a
crucial role for {OPTN} (optineurin) in acute myeloid
leukemia.},
journal = {Autophagy},
volume = {19},
number = {11},
issn = {1554-8627},
address = {Abingdon, Oxon},
publisher = {Taylor $\&$ Francis},
reportid = {DKFZ-2023-02006},
pages = {2982 - 2996},
year = {2023},
abstract = {The selective autophagic degradation of mitochondria via
mitophagy is essential for preserving mitochondrial
homeostasis and, thereby, disease maintenance and
progression in acute myeloid leukemia (AML). Mitophagy is
orchestrated by a variety of mitophagy receptors whose
interplay is not well understood. Here, we established a
pairwise multiplexed CRISPR screen targeting mitophagy
receptors to elucidate redundancies and gain a deeper
understanding of the functional interactome governing
mitophagy in AML. We identified OPTN (optineurin) as sole
non-redundant mitophagy receptor and characterized its
unique role in AML. Knockdown and overexpression experiments
demonstrated that OPTN expression is rate-limiting for AML
cell proliferation. In a MN1-driven murine transplantation
model, loss of OPTN prolonged overall median survival by 7
days $(+21\%).$ Mechanistically, we found broadly impaired
mitochondrial respiration and function with increased
mitochondrial ROS, that most likely caused the proliferation
defect. Our results decipher the intertwined network of
mitophagy receptors in AML for both ubiquitin-dependent and
receptor-mediated mitophagy, identify OPTN as a
non-redundant tool to study mitophagy in the context of
leukemia and suggest OPTN inhibition as an attractive
therapeutic strategy.Abbreviations: AML: acute myeloid
leukemia; CRISPR: Clustered Regularly Interspaced Short
Palindromic Repeats; CTRL: control; DFP: deferiprone; GI:
genetic interaction; KD: knockdown; KO: knockout; ldMBM,
lineage-depleted murine bone marrow; LFC: log2 fold change;
LIR: LC3-interacting region; LSC: leukemic stem cell;
MAGeCK: Model-based Analysis of Genome-wide CRISPR-Cas9
Knockout; MDIVI-1: mitochondrial division inhibitor 1; MOI:
multiplicity of infection; MOM: mitochondrial outer
membrane; NAC: N-acetyl-L-cysteine; OA: oligomycin-antimycin
A; OCR: oxygen consumption rate; OE: overexpression; OPTN:
optineurin; PINK1: PTEN induced putative kinase 1; ROS:
reactive oxygen species; SEM: standard error of the mean;
TCGA: The Cancer Genome Atlas; TEM: transmission electron
microscopy; UBD: ubiquitin-binding domain; WT: wild type.},
keywords = {Animals / Mice / Mitophagy: genetics / Reactive Oxygen
Species: metabolism / Autophagy / Leukemia, Myeloid, Acute /
Ubiquitins / Ubiquitin-Protein Ligases: metabolism / AML
(Other) / MN1-driven mouse model (Other) / Mitochondrial ROS
(Other) / Multiplex CRISPR screen (Other) / genetic
interactions (Other) / leukemia (Other) / Reactive Oxygen
Species (NLM Chemicals) / Mdivi-1 (NLM Chemicals) /
Ubiquitins (NLM Chemicals) / Ubiquitin-Protein Ligases (NLM
Chemicals)},
cin = {FM01},
ddc = {570},
cid = {I:(DE-He78)FM01-20160331},
pnm = {899 - ohne Topic (POF4-899)},
pid = {G:(DE-HGF)POF4-899},
typ = {PUB:(DE-HGF)16},
pubmed = {pmid:37439113},
doi = {10.1080/15548627.2023.2230839},
url = {https://inrepo02.dkfz.de/record/284417},
}