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BLOC-1 Brings Together the Actin and Microtubule Cytoskeletons to Generate Recycling Endosomes.

Published on Jan 1, 2016in Current Biology 9.19
· DOI :10.1016/j.cub.2015.11.020
Cédric Delevoye15
Estimated H-index: 15
(PSL Research University),
Xavier Heiligenstein4
Estimated H-index: 4
(PSL Research University)
+ 9 AuthorsGraça Raposo92
Estimated H-index: 92
(PSL Research University)
Cite
Abstract
Recycling endosomes consist of a tubular network that emerges from vacuolar sorting endosomes and diverts cargoes toward the cell surface, the Golgi, or lysosome-related organelles. How recycling tubules are formed remains unknown. We show that recycling endosome biogenesis requires the protein complex BLOC-1. Mutations in BLOC-1 subunits underlie an inherited disorder characterized by albinism, the Hermansky-Pudlak Syndrome, and are associated with schizophrenia risk. We show here that BLOC-1 coordinates the kinesin KIF13A-dependent pulling of endosomal tubules along microtubules to the Annexin A2/actin-dependent stabilization and detachment of recycling tubules. These components cooperate to extend, stabilize and form tubular endosomal carriers that function in cargo recycling and in the biogenesis of pigment granules in melanocytic cells. By shaping recycling endosomal tubules, our data reveal that dysfunction of the BLOC-1-KIF13A-Annexin A2 molecular network underlies the pathophysiology of neurological and pigmentary disorders.
  • References (48)
  • Citations (245)
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References48
Newest
Published on Nov 1, 2015in Nature Communications 11.88
Kevin Moreau19
Estimated H-index: 19
,
Ghita Ghislat8
Estimated H-index: 8
+ 9 AuthorsFiona M. Menzies32
Estimated H-index: 32
Autophagy is an important degradation pathway, which is induced after starvation, where it buffers nutrient deprivation by recycling macromolecules in organisms from yeast to man. While the classical pathway mediating this response is via mTOR inhibition, there are likely to be additional pathways that support the process. Here, we identify Annexin A2 as an autophagy modulator that regulates autophagosome formation by enabling appropriate ATG9A trafficking from endosomes to autophagosomes via ac...
22 Citations Source Cite
Published on May 25, 2015in Journal of Cell Biology 8.89
Megan K. Dennis12
Estimated H-index: 12
(UPenn: University of Pennsylvania),
Adriana R. Mantegazza5
Estimated H-index: 5
(UPenn: University of Pennsylvania)
+ 13 AuthorsKeerthana Ravichandran2
Estimated H-index: 2
(IISc: Indian Institute of Science)
Hermansky-Pudlak syndrome (HPS) is a group of disorders characterized by the malformation of lysosome-related organelles, such as pigment cell melanosomes. Three of nine characterized HPS subtypes result from mutations in subunits of BLOC-2, a protein complex with no known molecular function. In this paper, we exploit melanocytes from mouse HPS models to place BLOC-2 within a cargo transport pathway from recycling endosomal domains to maturing melanosomes. In BLOC-2-deficient melanocytes, the me...
21 Citations Source Cite
Published on Apr 1, 2015in Developmental Cell 9.19
Jing Pu14
Estimated H-index: 14
(NIH: National Institutes of Health),
Christina Schindler7
Estimated H-index: 7
(NIH: National Institutes of Health)
+ 3 AuthorsJuan S. Bonifacino92
Estimated H-index: 92
(NIH: National Institutes of Health)
Summary The positioning of lysosomes within the cytoplasm is emerging as a critical determinant of many lysosomal functions. Here we report the identification of a multisubunit complex named BORC that regulates lysosome positioning. BORC comprises eight subunits, some of which are shared with the BLOC-1 complex involved in the biogenesis of lysosome-related organelles, and the others of which are products of previously uncharacterized open reading frames. BORC associates peripherally with the ly...
99 Citations Source Cite
Published on Oct 1, 2014in Cold Spring Harbor Perspectives in Biology 9.11
Judith Klumperman62
Estimated H-index: 62
(UU: Utrecht University),
Graça Raposo92
Estimated H-index: 92
(Curie Institute)
Live-cell imaging reveals the endolysosomal system as a complex and highly dynamic network of interacting compartments. Distinct types of endosomes are discerned by kinetic, molecular, and morphological criteria. Although none of these criteria, or combinations thereof, can capture the full complexity of the endolysosomal system, they are extremely useful for experimental purposes. Some membrane domain specializations and specific morphological characteristics can only be seen by ultrastructural...
81 Citations Source Cite
Published on Jul 1, 2014in Seminars in Cell & Developmental Biology 5.46
Elizabeth Granger1
Estimated H-index: 1
(University of Manchester),
Gavin McNee1
Estimated H-index: 1
(University of Manchester)
+ 1 AuthorsPhilip Woodman31
Estimated H-index: 31
(University of Manchester)
The endocytic pathway is essential for processes that define how cells interact with their environment, including receptor signalling, cell adhesion and migration, pathogen entry, membrane protein turnover and nutrient uptake. The spatial organisation of endocytic trafficking requires motor proteins that tether membranes or transport them along the actin and microtubule cytoskeletons. Microtubules, actin filaments and motor proteins also provide force to deform and assist in the scission of memb...
73 Citations Source Cite
Published on Jun 1, 2014in Traffic 3.95
Xavier Heiligenstein4
Estimated H-index: 4
(Curie Institute),
Jérôme Heiligenstein1
Estimated H-index: 1
(CNRS: Centre national de la recherche scientifique)
+ 8 AuthorsClaude Antony2
Estimated H-index: 2
(French Institute of Health and Medical Research)
Correlating complementary multiple scale images of the same object is a straightforward means to decipher biological processes. Light microscopy and electron microscopy are the most commonly used imaging techniques, yet despite their complementarity, the experimental procedures available to correlate them are technically complex. We designed and manufactured a new device adapted to many biological specimens, the CryoCapsule, that simplifies the multiple sample preparation steps, which at present...
16 Citations Source Cite
Published on Feb 1, 2014in Cell Reports 7.82
Cédric Delevoye15
Estimated H-index: 15
(Curie Institute),
Stéphanie Miserey-Lenkei10
Estimated H-index: 10
(Curie Institute)
+ 7 AuthorsGraça Raposo92
Estimated H-index: 92
(Curie Institute)
Present address: Membrane Dynamics and Intracellular Trafficking, Institut Jacques Monod, UMR 7592, CNRS, Universite´ Paris Diderot,Sorbonne Paris Cite´, Paris 75013, France*Correspondence: cedric.delevoye@curie.frhttp://dx.doi.org/10.1016/j.celrep.2014.01.002This is an open-access article distributed under the terms of the Creative Commons Attribution-NonCommercial-No Derivative WorksLicense, which permits non-commercial use, distribution, and reproduction in any medium, provided the original au...
56 Citations Source Cite
Published on Jan 1, 2014in Methods in Cell Biology 1.70
Xavier Heiligenstein4
Estimated H-index: 4
(Curie Institute),
Ilse Hurbain20
Estimated H-index: 20
(Curie Institute)
+ 3 AuthorsGraça Raposo92
Estimated H-index: 92
(Curie Institute)
Abstract The CryoCapsule is a tool dedicated to correlative light to electron microscopy experiments. Focused on simplifying the specimen manipulation throughout the entire workflow from live-cell imaging to freeze substitution following cryofixation by high pressure freezing, we introduce here a step by step procedure to use the CryoCapsule either with the high pressure freezing machines: HPM010 or the HPM100.
4 Citations Source Cite
Published on Aug 1, 2013in Current Opinion in Cell Biology 8.23
Michael S. Marks51
Estimated H-index: 51
(UPenn: University of Pennsylvania),
Harry F. G. Heijnen29
Estimated H-index: 29
(French Institute of Health and Medical Research),
Graça Raposo92
Estimated H-index: 92
(Curie Institute)
Lysosome-related organelles (LROs) comprise a group of cell type-specific subcellular compartments with unique composition, morphology and structure that share some features with endosomes and lysosomes and that function in varied processes such as pigmentation, hemostasis, lung plasticity and immunity. In recent years, studies of genetic diseases in which LRO functions are compromised have provided new insights into the mechanisms of LRO biogenesis and the regulated secretion of LRO contents. T...
121 Citations Source Cite
Published on Jul 15, 2013in Molecular Biology of the Cell 3.90
Pearl V. Ryder6
Estimated H-index: 6
(Emory University),
Rachel Vistein6
Estimated H-index: 6
(CMU: Carnegie Mellon University)
+ 3 AuthorsVictor Faundez39
Estimated H-index: 39
(Emory University)
Vesicle biogenesis machinery components such as coat proteins can interact with the actin cytoskeleton for cargo sorting into multiple pathways. It is unknown, however, whether these interactions are a general requirement for the diverse endosome traffic routes. In this study, we identify actin cytoskeleton regulators as previously unrecognized interactors of complexes associated with the Hermansky–Pudlak syndrome. Two complexes mutated in the Hermansky–Pudlak syndrome, adaptor protein complex-3...
41 Citations Source Cite
Cited By245
Newest
Published on Feb 18, 2019in Nature Communications 11.88
Lusha Zhu5
Estimated H-index: 5
(McGovern Institute for Brain Research),
Yaomin Jiang (McGovern Institute for Brain Research)+ 2 AuthorsMing Hsu15
Estimated H-index: 15
(University of California, Berkeley)
Both basal ganglia (BG) and orbitofrontal cortex (OFC) have been widely implicated in social and non-social decision-making. However, unlike OFC damage, BG pathology is not typically associated with disturbances in social functioning. Here we studied the behavior of patients with focal lesions to either BG or OFC in a multi-strategy competitive game known to engage these regions. We find that whereas OFC patients are significantly impaired, BG patients show intact learning in the economic game. ...
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Published on Mar 4, 2019in BMC Evolutionary Biology 3.04
Tatiana Aghová5
Estimated H-index: 5
,
Klára Palupčíková (Charles University in Prague)+ 8 AuthorsDavid Modrý28
Estimated H-index: 28
Background Spiny mice of the genus Acomys are distributed mainly in dry open habitats in Africa and the Middle East, and they are widely used as model taxa for various biological disciplines (e.g. ecology, physiology and evolutionary biology). Despite their importance, large distribution and abundance in local communities, the phylogeny and the species limits in the genus are poorly resolved, and this is especially true for sub-Saharan taxa. The main aims of this study are (1) to reconstruct phy...
1 Citations Source Cite
Published on Aug 1, 2019in Current Opinion in Cell Biology 8.23
Cortnie Hartwig4
Estimated H-index: 4
(Emory University),
Stephanie A. Zlatic11
Estimated H-index: 11
(Emory University)
+ 3 AuthorsVictor Faundez39
Estimated H-index: 39
(Emory University)
Copper is an essential micronutrient required for oxygen-dependent enzymes, yet excess of the metal is a toxicant. The tug-of-war between these copper activities is balanced by chaperones and membrane transporters, which control copper distribution and availability. The P-type ATPase transporters, ATP7A and ATP7B, regulate cytoplasmic copper by pumping copper out of cells or into the endomembrane system. Mutations in ATP7A and ATP7B cause diseases that share neuropsychiatric phenotypes, which ar...
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Published on Aug 1, 2019in Current Opinion in Cell Biology 8.23
Cédric Delevoye15
Estimated H-index: 15
(CNRS: Centre national de la recherche scientifique),
Michael S. Marks51
Estimated H-index: 51
(UPenn: University of Pennsylvania),
Graça Raposo92
Estimated H-index: 92
(CNRS: Centre national de la recherche scientifique)
Unique functions of specialised cells such as those of the immune and haemostasis systems, skin, blood vessels, lung, and bone require specialised compartments, collectively referred to as lysosome-related organelles (LROs), that share features of endosomes and lysosomes. LROs harbour unique morphological features and cell type-specific contents, and most if not all undergo regulated secretion for diverse functions. Ongoing research, largely driven by analyses of inherited diseases and their mod...
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Published on Jul 8, 2019in Nature Human Behaviour
Chunyue Teng (GW: George Washington University), Dwight J. Kravitz20
Estimated H-index: 20
(GW: George Washington University)
Visual working memory (VWM), the ability to temporarily maintain and manipulate information, underlies a variety of critical high-level behaviours from directing attention1–4 to making complex decisions5. Here we show that its impact extends to even the most basic levels of perceptual processing, directly interacting with and even distorting the physical appearance of visual features. This interference results from and can be predicted by the recruitment of posterior perceptual cortices to maint...
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Published on Jan 23, 2019in Protoplasma 2.63
Tengfei Ma1
Estimated H-index: 1
(WHU: Wuhan University),
Fang Dong (WHU: Wuhan University)+ 2 AuthorsJie Zhao11
Estimated H-index: 11
(WHU: Wuhan University)
Arabinogalactan proteins (AGPs) are hyperglycosylated members of the hydroxyproline-rich glycoprotein (HRGP) superfamily and are widely distributed throughout the plant kingdom. In Oryza sativa (rice), the gene expression and biological function of AGPs only have received minimal research attention. Here, we used qRT-PCR to detect the expression patterns of OsAGP genes in various organs, and found that six genes were preferentially expressed in panicles, three genes were specifically expressed i...
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