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ATG9A and ARFIP2 cooperate to control PI4P levels for lysosomal repair

  • Stefano De Tito*
  • , Eugenia Almacellas
  • , Daniel Dai Yu
  • , Emily Millard
  • , Wenxin Zhang
  • , Cecilia de Heus
  • , Christophe Queval
  • , Javier H Hervás
  • , Enrica Pellegrino
  • , Ioanna Panagi
  • , Ditte Fogde
  • , Teresa L M Thurston
  • , Judith Klumperman
  • , Maximiliano Gutierrez
  • , Sharon A Tooze*
  • *Corresponding author for this work

Research output: Contribution to journalArticleAcademicpeer-review

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Abstract

Lysosome damage activates multiple pathways to prevent lysosome-dependent cell death, including a repair mechanism involving endoplasmic reticulum (ER)-lysosome membrane contact sites, phosphatidylinositol 4-kinase-2a (PI4K2A), phosphatidylinositol-4 phosphate (PI4P), and oxysterol-binding protein-like proteins (OSBPLs) lipid transfer proteins. PI4K2A localizes to the trans-Golgi network and endosomes, yet how it is delivered to damaged lysosomes remains unknown. During acute sterile damage and damage caused by intracellular bacteria, we show that ATG9A-containing vesicles perform a critical role in delivering PI4K2A to damaged lysosomes. ADP ribosylation factor interacting protein 2 (ARFIP2), a component of ATG9A vesicles, binds and sequesters PI4P on lysosomes, balancing OSBPL-dependent lipid transfer and promoting the retrieval of ATG9A vesicles through the recruitment of the adaptor protein complex-3 (AP-3). Our results identify a role for mobilized ATG9A vesicles and ARFIP2 in lysosome homeostasis after damage and bacterial infection.

Original languageEnglish
Pages (from-to)2744-2760.e9
JournalDevelopmental Cell
Volume60
Issue number20
Early online date27 May 2025
DOIs
Publication statusPublished - 20 Oct 2025

Keywords

  • AP-3
  • ARFIP2
  • ATG9A
  • PI4K2A
  • PI4P
  • autophagy
  • lysosomal damage
  • lysosome
  • membrane trafficking

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