Atp6ap2 deletion causes extensive vacuolation that consumes the insulin content of pancreatic β cells

Research output: Contribution to journalResearch articleContributedpeer-review

Contributors

  • Katrina J Binger - , La Trobe University (Author)
  • Martin Neukam - , Molecular Diabetology, TUD Dresden University of Technology, University Hospital Carl Gustav Carus Dresden (Author)
  • Sudhir Gopal Tattikota - , Massachusetts General Hospital (Author)
  • Fatimunnisa Qadri - , Max Delbrück Center for Molecular Medicine (MDC) (Author)
  • Dmytro Puchkov - , Leibniz Institute for Molecular Pharmacology (Author)
  • Diana M Willmes - , University Hospital Carl Gustav Carus Dresden (Author)
  • Sabrina Wurmsee - , Max Delbrück Center for Molecular Medicine (MDC) (Author)
  • Sabrina Geisberger - , Max Delbrück Center for Molecular Medicine (MDC) (Author)
  • Ralf Dechend - , Experimental and Clinical Research Center (ECRC) (Author)
  • Klemens Raile - , Experimental and Clinical Research Center (ECRC) (Author)
  • Thomas Kurth - , Core Facility Electron Microscopy & Histology, Center for Molecular and Cellular Bioengineering (CMCB), Center for Regenerative Therapies Dresden (Author)
  • Genevieve Nguyen - , Center for Interdisciplinary Research in Biology (Author)
  • Matthew N Poy - , Max Delbrück Center for Molecular Medicine (MDC) (Author)
  • Michele Solimena - , Molecular Diabetology, TUD Dresden University of Technology, University Hospital Carl Gustav Carus Dresden (Author)
  • Dominik N Muller - , Max Delbrück Center for Molecular Medicine (MDC) (Author)
  • Andreas L Birkenfeld - , Molecular Diabetology, TUD Dresden University of Technology, University Hospital Carl Gustav Carus Dresden, University Hospital Tübingen, Helmholtz Zentrum München - German Research Center for Environmental Health, University of Tübingen, King's College London (KCL) (Author)

Abstract

Pancreatic β cells store insulin within secretory granules which undergo exocytosis upon elevation of blood glucose levels. Crinophagy and autophagy are instead responsible to deliver damaged or old granules to acidic lysosomes for intracellular degradation. However, excessive consumption of insulin granules can impair β cell function and cause diabetes. Atp6ap2 is an essential accessory component of the vacuolar ATPase required for lysosomal degradative functions and autophagy. Here, we show that Cre recombinase-mediated conditional deletion of Atp6ap2 in mouse β cells causes a dramatic accumulation of large, multigranular vacuoles in the cytoplasm, with reduction of insulin content and compromised glucose homeostasis. Loss of insulin stores and gigantic vacuoles were also observed in cultured insulinoma INS-1 cells upon CRISPR/Cas9-mediated removal of Atp6ap2. Remarkably, these phenotypic alterations could not be attributed to a deficiency in autophagy or acidification of lysosomes. Together, these data indicate that Atp6ap2 is critical for regulating the stored insulin pool and that a balanced regulation of granule turnover is key to maintaining β cell function and diabetes prevention.

Details

Original languageEnglish
Pages (from-to)19983-19988
Number of pages6
JournalProceedings of the National Academy of Sciences of the United States of America : PNAS
Volume116
Issue number40
Publication statusPublished - 1 Oct 2019
Peer-reviewedYes

External IDs

PubMedCentral PMC6778207
Scopus 85072758913
ORCID /0000-0001-5624-1717/work/142239057

Keywords

Sustainable Development Goals

Keywords

  • Animals, Autophagy, CRISPR-Cas Systems, Cytosol/metabolism, Female, Gene Deletion, Gene Silencing, Insulin/metabolism, Insulin-Secreting Cells/metabolism, Insulinoma/metabolism, Lysosomes/metabolism, Male, Mice, Phenotype, Promoter Regions, Genetic, Proton-Translocating ATPases/genetics, RNA, Small Interfering/metabolism, Rats, Receptors, Cell Surface/genetics, Receptors, Estrogen/metabolism, Vacuolar Proton-Translocating ATPases/metabolism, Vacuoles/metabolism