VPS13D bridges the ER to mitochondria and peroxisomes via Miro

VPS13D mutations result in severe mitochondrial defects. Guillén-Samander et al. show that VPS13D binds VAP in the ER and interacts with Miro on mitochondria and peroxisomes, where it could provide a bridge for lipid transport between these organelles.

[1]  N. Brüggemann,et al.  VPS13D promotes peroxisome biogenesis , 2021, Journal of Cell Biology.

[2]  J. Shaw,et al.  Amino Acids Promote Mitochondrial-Derived Compartment Formation in Mammalian Cells , 2020, bioRxiv.

[3]  G. Drin,et al.  FFAT motif phosphorylation controls formation and lipid transfer function of inter‐organelle contacts , 2020, The EMBO journal.

[4]  J. Shaw,et al.  ER–mitochondria contacts promote mitochondrial-derived compartment biogenesis , 2020, The Journal of cell biology.

[5]  A. Neiman,et al.  XK is a partner for VPS13A: a molecular link between Chorea-Acanthocytosis and McLeod Syndrome , 2020, Molecular biology of the cell.

[6]  P. De Camilli,et al.  Role of VPS13, a protein with similarity to ATG2, in physiology and disease. , 2020, Current opinion in genetics & development.

[7]  E. Shoubridge,et al.  A high-density human mitochondrial proximity interaction network , 2020, bioRxiv.

[8]  J. Lees,et al.  Inter-organelle lipid transfer: a channel model for Vps13 and chorein-N motif proteins. , 2020, Current opinion in cell biology.

[9]  C. Lusk,et al.  Cryo-EM reconstruction of a VPS13 fragment reveals a long groove to channel lipids between membranes , 2020, The Journal of cell biology.

[10]  Catherine A. Collins,et al.  Vps13D is required for mitochondrial fission and mitophagy triggered by fission defects in Drosophila neurons , 2020, bioRxiv.

[11]  Michael Davey,et al.  A VPS13D spastic ataxia mutation disrupts the conserved adaptor-binding site in yeast Vps13 , 2020, Human molecular genetics.

[12]  G. López-Doménech,et al.  Peroxisomal fission is modulated by the mitochondrial Rho‐GTPases, Miro1 and Miro2 , 2020, EMBO reports.

[13]  R. Krüger,et al.  Variants in Miro1 Cause Alterations of ER-Mitochondria Contact Sites in Fibroblasts from Parkinson’s Disease Patients , 2019, Journal of clinical medicine.

[14]  Alan R. Lowe,et al.  Miro clusters regulate ER-mitochondria contact sites and link cristae organization to the mitochondrial transport machinery , 2019, Nature Communications.

[15]  J. Lippincott-Schwartz,et al.  ER membranes exhibit phase behavior at sites of organelle contact , 2019, Proceedings of the National Academy of Sciences.

[16]  T. Walz,et al.  ATG2 transports lipids to promote autophagosome biogenesis , 2019, The Journal of cell biology.

[17]  B. Kornmann,et al.  Lipid exchange at ER-mitochondria contact sites: a puzzle falling into place with quite a few pieces missing. , 2019, Current opinion in cell biology.

[18]  P. De Camilli,et al.  Lipid transporter TMEM24/C2CD2L is a Ca2+-regulated component of ER–plasma membrane contacts in mammalian neurons , 2019, Proceedings of the National Academy of Sciences.

[19]  T. Otomo,et al.  The autophagic membrane tether ATG2A transfers lipids between membranes , 2019, bioRxiv.

[20]  A. Monaco,et al.  Human VPS13A is associated with multiple organelles and influences mitochondrial morphology and lipid droplet motility , 2019, eLife.

[21]  J. Slee,et al.  Systematic Prediction of FFAT Motifs Across Eukaryote Proteomes Identifies Nucleolar and Eisosome Proteins With the Predicted Capacity to Form Bridges to the Endoplasmic Reticulum , 2019, Contact (Thousand Oaks (Ventura County, Calif.)).

[22]  I. J. van der Klei,et al.  Yeast peroxisomes: How are they formed and how do they grow? , 2018, The international journal of biochemistry & cell biology.

[23]  T. Levine,et al.  Lipid transfer proteins: the lipid commute via shuttles, bridges and tubes , 2018, Nature Reviews Molecular Cell Biology.

[24]  M. Bähler,et al.  Identification of Miro1 and Miro2 as mitochondrial receptors for myosin XIX , 2018, Journal of Cell Science.

[25]  P. De Camilli,et al.  VPS13A and VPS13C are lipid transport proteins differentially localized at ER contact sites , 2018, The Journal of cell biology.

[26]  Michael Davey,et al.  Competitive organelle-specific adaptors recruit Vps13 to membrane contact sites , 2018, The Journal of cell biology.

[27]  J. Trimmer,et al.  Identification of VAPA and VAPB as Kv2 Channel-Interacting Proteins Defining Endoplasmic Reticulum–Plasma Membrane Junctions in Mammalian Brain Neurons , 2018, The Journal of Neuroscience.

[28]  M. Tamkun,et al.  Kv2 potassium channels form endoplasmic reticulum/plasma membrane junctions via interaction with VAPA and VAPB , 2018, Proceedings of the National Academy of Sciences.

[29]  Jun Z. Li,et al.  Mutations in VPS13D lead to a new recessive ataxia with spasticity and mitochondrial defects , 2018, Annals of neurology.

[30]  H. Prokisch,et al.  Recessive mutations in VPS13D cause childhood onset movement disorders , 2018, Annals of neurology.

[31]  Xiaonan Liu,et al.  An AP-MS- and BioID-compatible MAC-tag enables comprehensive mapping of protein interactions and subcellular localizations , 2018, Nature Communications.

[32]  M. Schrader,et al.  A role for Mitochondrial Rho GTPase 1 (MIRO1) in motility and membrane dynamics of peroxisomes , 2018, Traffic.

[33]  K. Okumoto,et al.  New splicing variants of mitochondrial Rho GTPase-1 (Miro1) transport peroxisomes , 2018, The Journal of cell biology.

[34]  T. Strom,et al.  Diagnostic exome sequencing in early‐onset Parkinson's disease confirms VPS13C as a rare cause of autosomal‐recessive Parkinson's disease , 2018, Clinical genetics.

[35]  Keiji Tanaka,et al.  Endosomal Rab cycles regulate Parkin-mediated mitophagy , 2018, eLife.

[36]  K. Hofmann,et al.  Vps13D Encodes a Ubiquitin-Binding Protein that Is Required for the Regulation of Mitochondrial Size and Clearance , 2018, Current Biology.

[37]  G. López-Doménech,et al.  Miro proteins coordinate microtubule‐ and actin‐dependent mitochondrial transport and distribution , 2018, The EMBO journal.

[38]  K. Dimmer,et al.  Vps13-Mcp1 interact at vacuole–mitochondria interfaces and bypass ER–mitochondria contact sites , 2017, The Journal of cell biology.

[39]  P. De Camilli,et al.  Endoplasmic Reticulum-Plasma Membrane Contact Sites. , 2017, Annual review of biochemistry.

[40]  Stephanie S. Lam,et al.  Proteomic mapping of cytosol-facing outer mitochondrial and ER membranes in living human cells by proximity biotinylation , 2017, eLife.

[41]  T. Schwarz,et al.  Miro phosphorylation sites regulate Parkin recruitment and mitochondrial motility , 2016, Proceedings of the National Academy of Sciences.

[42]  T. Levine,et al.  VAP, a Versatile Access Point for the Endoplasmic Reticulum: Review and analysis of FFAT-like motifs in the VAPome. , 2016, Biochimica et biophysica acta.

[43]  A. Neiman,et al.  Yeast Vps13 promotes mitochondrial function and is localized at membrane contact sites , 2016, Molecular biology of the cell.

[44]  P. De Camilli,et al.  Endosome-ER Contacts Control Actin Nucleation and Retromer Function through VAP-Dependent Regulation of PI4P , 2016, Cell.

[45]  Simon C. Potter,et al.  Loss of VPS13C Function in Autosomal-Recessive Parkinsonism Causes Mitochondrial Dysfunction and Increases PINK1/Parkin-Dependent Mitophagy. , 2016, American journal of human genetics.

[46]  G. Superti-Furga,et al.  Gene essentiality and synthetic lethality in haploid human cells , 2015, Science.

[47]  E. Lander,et al.  Identification and characterization of essential genes in the human genome , 2015, Science.

[48]  P. Walter,et al.  ER-mitochondrial junctions can be bypassed by dominant mutations in the endosomal protein Vps13. , 2015, The Journal of cell biology.

[49]  M. Peter,et al.  Mitotic redistribution of the mitochondrial network by Miro and Cenp-F , 2015, Nature Communications.

[50]  Brian Kuhlman,et al.  Engineering an improved light-induced dimer (iLID) for controlling the localization and activity of signaling proteins , 2014, Proceedings of the National Academy of Sciences.

[51]  D. Horn,et al.  Cohen Syndrome-associated Protein COH1 Physically and Functionally Interacts with the Small GTPase RAB6 at the Golgi Complex and Directs Neurite Outgrowth* , 2014, The Journal of Biological Chemistry.

[52]  S. Pulst,et al.  Loss of Miro1-directed mitochondrial movement results in a novel murine model for neuron disease , 2014, Proceedings of the National Academy of Sciences.

[53]  W. Prinz.  Bridging the gap: Membrane contact sites in signaling, metabolism, and organelle dynamics , 2014, The Journal of cell biology.

[54]  David Komander,et al.  Lysine 27 Ubiquitination of the Mitochondrial Transport Protein Miro Is Dependent on Serine 65 of the Parkin Ubiquitin Ligase* , 2014, The Journal of Biological Chemistry.

[55]  T. Langer,et al.  Mitochondrial lipid transport at a glance , 2013, Journal of Cell Science.

[56]  M. Schrader,et al.  The peroxisome: an update on mysteries 2.0 , 2012, Histochemistry and Cell Biology.

[57]  Xinnan Wang,et al.  PINK1 and Parkin Target Miro for Phosphorylation and Degradation to Arrest Mitochondrial Motility , 2011, Cell.

[58]  D. Horn,et al.  Cohen Syndrome-associated Protein, COH1, Is a Novel, Giant Golgi Matrix Protein Required for Golgi Integrity* , 2011, The Journal of Biological Chemistry.

[59]  P. Walter,et al.  The conserved GTPase Gem1 regulates endoplasmic reticulum–mitochondria connections , 2011, Proceedings of the National Academy of Sciences.

[60]  Atsushi Tanaka,et al.  PINK1 Is Selectively Stabilized on Impaired Mitochondria to Activate Parkin , 2010, PLoS biology.

[61]  Peter Walter,et al.  Supporting Online Material for An ER-Mitochondria Tethering Complex Revealed by a Synthetic Biology Screen , 2009 .

[62]  D. Attwell,et al.  Miro1 Is a Calcium Sensor for Glutamate Receptor-Dependent Localization of Mitochondria at Synapses , 2009, Neuron.

[63]  Xinnan Wang,et al.  The Mechanism of Ca2+-Dependent Regulation of Kinesin-Mediated Mitochondrial Motility , 2009, Cell.

[64]  G. Hajnóczky,et al.  Bidirectional Ca2+-dependent control of mitochondrial dynamics by the Miro GTPase , 2008, Proceedings of the National Academy of Sciences.

[65]  W. Prinz,et al.  Nonvesicular phospholipid transfer between peroxisomes and the endoplasmic reticulum , 2008, Proceedings of the National Academy of Sciences.

[66]  T. Schwarz,et al.  Axonal transport of mitochondria requires milton to recruit kinesin heavy chain and is light chain independent , 2006, The Journal of cell biology.

[67]  Rebecca L. Frederick,et al.  Yeast Miro GTPase, Gem1p, regulates mitochondrial morphology via a novel pathway , 2004, The Journal of cell biology.

[68]  A. Monaco,et al.  Analysis of the human VPS13 gene family. , 2004, Genomics.

[69]  S. Gould,et al.  PEX19 is a predominantly cytosolic chaperone and import receptor for class 1 peroxisomal membrane proteins , 2004, The Journal of cell biology.

[70]  J. Clayton-Smith,et al.  Cohen syndrome is caused by mutations in a novel gene, COH1, encoding a transmembrane protein with a presumed role in vesicle-mediated sorting and intracellular protein transport. , 2003, American journal of human genetics.

[71]  A. Ruusala,et al.  Atypical Rho GTPases Have Roles in Mitochondrial Homeostasis and Apoptosis* , 2003, The Journal of Biological Chemistry.

[72]  Y. Maruki,et al.  The gene encoding a newly discovered protein, chorein, is mutated in chorea-acanthocytosis , 2001, Nature Genetics.

[73]  Adrian Danek,et al.  A conserved sorting-associated protein is mutant in chorea-acanthocytosis , 2001, Nature Genetics.

[74]  J. Vance.  Phospholipid synthesis in a membrane fraction associated with mitochondria. , 1990, The Journal of biological chemistry.

[75]  T. Osawa,et al.  Atg2 mediates direct lipid transfer between membranes for autophagosome formation , 2019, Nature Structural & Molecular Biology.

[76]  Arman Akşit.  Peroxisomal membrane contact sites in the yeast Hansenula polymorpha , 2018 .

[77]  Frauke Pohlki,et al.  The Mechanism of the , 2001, Angewandte Chemie.