Acknowledgements: We thank members of the Arnesen, Martinho, Boone, and Andrews labs for helpful discussions. Lena Thurnes, Kjellfrid Haukenes, Nina Glomnes, Olga Sizova, and Andrea Habsid are gratefully acknowledged for technical assistance. We also thank Dr. Jaakko Saraste and Dr. Marc Niere for help and advice. Furthermore, we thank Dr. Scott J. Dixon and Dr. Stian Knappskog for cell lines, Dr. Eileen Furlong for antibody, as well as Dr. Yong Tae Kwon, Dr. Fabio Demontis, and Dr. Liam C. Hunt for kindly providing plasmids. We also thank Dr. Paulo Navarro-Costa for his contribution to the Drosophila testis analysis. Next-generations sequencing was performed at the Centre for Applied Genomics, The Hospital for Sick Children, Toronto, Canada. Proteomics work of human cells lines was performed at the VIB Proteomics Core, located at the VIB-UGent Center for Medical Biotechnology, Belgium, and was supported by EPIC-XS, project number 823839, funded by the Horizon 2020 programme of the European Union. Proteomics work of Drosophila purified proteins was performed at the Mass Spectrometry Laboratory, Institute of Biochemistry and Biophysics, Polish Academy of Sciences, Warsaw, Poland. Sequencing was performed at Haukeland University Hospital. Flow cytometry analysis and cell sorting were performed at the Flow Cytometry Core Facility, Department of Clinical Science, University of Bergen, Norway, with advice from Dr. Brith Bergum. Confocal imaging was performed at the Molecular Imaging Center (MIC), Department of Biomedicine, University of Bergen. This work was supported by grants from the Research Council of Norway (RCN) (FRIPRO Mobility Grant 261981 to S.V. which was co-funded by the European Union’s Seventh Framework Programme under Marie Curie grant agreement no 608695, and FRIPRO grant 249843 to T.A.), the Norwegian Health Authorities of Western Norway (F-12540 to T.A.), the Norwegian Cancer Society (171752-PR-2009-0222 to T.A), the European Research Council (ERC) under the European Union Horizon 2020 Research and Innovation Program (Grant 772039 to T.A), the Research Foundation - Flanders (FWO) (G008018N and G002721N to K.G.), the French National Centre for Scientific Research (CNRS) (ATIP-Avenir grant to H.B), AFM-Telethon (Trampoline grant 23108 to H.B.), the Medical Research Council (MRC) (MC_UU_00028/6 to A.J.W), the Canadian Institutes of Health Research (CIHR) (FDN-143264 and FDN-143265 to C.B. and B.J.A.; PJT-180285 to C.B, PJT-463531 to J.M.), the National Institutes of Health (NIH) (R01HG005853 to B.J.A., C.B., and C.L.M. R01HG005084 to C.L.M.), and the Portuguese national funding through Fundação para a Ciência e a Tecnologia (FCT) (DL 57/2016/CP1361/CT0019 and 2022.01782.PTDC to R.D.S; PTDC/BIA-BID/28441/2017 and PTDC/BIA-BID/1606/2020 to R.G.M). This particular work was partially funded by Algarve Regional Operational Program - CRESC Algarve 2020 and by National Funds through Fundação para a Ciência e a Tecnologia (FCT), under the project ALG-01-0145-FEDER-028441. The Algarve Biomedical Center Research Institute Microscopy Unit was partially supported by the Portuguese national funding (PPBI-POCI-01-0145-FEDER-022122). This work was developed with the support of the research infrastructure Congento (LISBOA-01-0145-FEDER-022170). The funding bodies had no roles in study design, data collection, data analysis, data interpretation, or manuscript writing.

Funder: U.S. Department of Health & Human Services | NIH | Office of Extramural Research, National Institutes of Health (OER)