ArticleThe Journal of cell biology2015
BLOC-2 targets recycling endosomal tubules to melanosomes for cargo delivery.
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Article in The Journal of cell biology, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 48 papers.
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48 citing papers in PubMed, 75 citations in OpenAlex.
- Flower Delivery: The Emergence of FLOWER (FWE) as a Multifunctional Regulator of Endo-Lysosome-Related Organelle Trafficking Across Cell Types.BioEssays : news and reviews in molecular, cellular and developmental biology · 2026Review
- Functional and Morphological Plasticity of the Endolysosomal System: Pigment Organelles at the Crossroads of Physiology and Pathology.Biology of the cell · 2025Review
- BLOC-1 and BORC: Complex regulators of endolysosomal dynamics.Cell chemical biology · 2025Review
- Genetics of Skin, Hair, and Eye Color in Human Pigmentation Disorders.Annals of human genetics · 2025Review
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- The clues offered by SNAREs on the vacuoles of plants and animals.Frontiers in plant science · 2025Review
- Insights into lysosome-related organelle biogenesis: melanosome as a model organelle.Frontiers in cell and developmental biology · 2025Article
- Evolutionary origins of the lysosome-related organelle sorting machinery reveal ancient homology in post-endosome trafficking pathways.Proceedings of the National Academy of Sciences of the United States of America · 2024Article
- LYSMD proteins promote activation of Rab32-family GTPases for lysosome-related organelle biogenesis.The Journal of cell biology · 2024Article
- Evoked Weibel-Palade Body Exocytosis Modifies the Endothelial Cell Surface by Releasing a Substrate-Selective Phosphodiesterase.Advanced science (Weinheim, Baden-Wurttemberg, Germany) · 2024Article
- Genetic architecture and evolution of color variation in American black bears.Current biology : CB · 2023Article
- Overlapping Machinery in Lysosome-Related Organelle Trafficking: A Lesson from Rare Multisystem Disorders.Cells · 2022Review
- Integrative modeling reveals the molecular architecture of the intraflagellar transport A (IFT-A) complex.eLife · 2022Article
- Type II phosphatidylinositol 4-kinases function sequentially in cargo delivery from early endosomes to melanosomes.The Journal of cell biology · 2022Article
- PI4P and BLOC-1 remodel endosomal membranes into tubules.The Journal of cell biology · 2022Article
- Review
- Zebrafish Syndromic Albinism Models as Tools for Understanding and Treating Pigment Cell Disease in Humans.Cancers · 2022Review
- Gene Expression Profile ofFrontiers in medicine · 2022Article
- Oculocutaneous albinism and bleeding diathesis due to a novel deletion in theFrontiers in genetics · 2022Article
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Authors and funding
16 authors at 6 institutions in 5 countries.
Megan K DennisDepartment of Pathology and Laboratory Medicine, Children's Hospital of Philadelphia, Philadelphia, PA 19104 Department of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104 Department of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104.
Adriana R MantegazzaDepartment of Pathology and Laboratory Medicine, Children's Hospital of Philadelphia, Philadelphia, PA 19104 Department of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104 Department of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104.
Olivia L SnirDepartment of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104 Department of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104.
Danièle TenzaInstitut Curie, Centre de Recherche; Structure and Membrane Compartments, Centre National de la Recherche Scientifique, Unité Mixte de Recherche (UMR) 144; and Cell and Tissue Imaging Facility, Centre National de la Recherche Scientifique UMR144, Paris F-75248, France Institut Curie, Centre de Recherche; Structure and Membrane Compartments, Centre National de la Recherche Scientifique, Unité Mixte de Recherche (UMR) 144; and Cell and Tissue Imaging Facility, Centre National de la Recherche Scientifique UMR144, Paris F-75248, France.
Amanda Acosta-RuizDepartment of Pathology and Laboratory Medicine, Children's Hospital of Philadelphia, Philadelphia, PA 19104 Department of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104 Department of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104.
Cédric DelevoyeInstitut Curie, Centre de Recherche; Structure and Membrane Compartments, Centre National de la Recherche Scientifique, Unité Mixte de Recherche (UMR) 144; and Cell and Tissue Imaging Facility, Centre National de la Recherche Scientifique UMR144, Paris F-75248, France Institut Curie, Centre de Recherche; Structure and Membrane Compartments, Centre National de la Recherche Scientifique, Unité Mixte de Recherche (UMR) 144; and Cell and Tissue Imaging Facility, Centre National de la Recherche Scientifique UMR144, Paris F-75248, France.
Richard ZorgerDepartment of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104.
Anand SitaramDepartment of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104 Department of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104.
Wilfredo de Jesus-RojasDepartment of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104 Department of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104.
Keerthana RavichandranDepartment of Microbiology and Cell Biology, Indian Institute of Science, Bangalore, India 560 012.
John RuxDepartment of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104 In Silico Molecular, LLC, Blue Bell, PA 19422.
Elena V SviderskayaMolecular Cell Sciences Research Centre, St. George's, University of London, London SW17 0RE, England, UK.
Dorothy C BennettMolecular Cell Sciences Research Centre, St. George's, University of London, London SW17 0RE, England, UK.
Graça RaposoInstitut Curie, Centre de Recherche; Structure and Membrane Compartments, Centre National de la Recherche Scientifique, Unité Mixte de Recherche (UMR) 144; and Cell and Tissue Imaging Facility, Centre National de la Recherche Scientifique UMR144, Paris F-75248, France Institut Curie, Centre de Recherche; Structure and Membrane Compartments, Centre National de la Recherche Scientifique, Unité Mixte de Recherche (UMR) 144; and Cell and Tissue Imaging Facility, Centre National de la Recherche Scientifique UMR144, Paris F-75248, France Institut Curie, Centre de Recherche; Structure and Membrane Compartments, Centre National de la Recherche Scientifique, Unité Mixte de Recherche (UMR) 144; and Cell and Tissue Imaging Facility, Centre National de la Recherche Scientifique UMR144, Paris F-75248, France.
Michael S MarksDepartment of Pathology and Laboratory Medicine, Children's Hospital of Philadelphia, Philadelphia, PA 19104 Department of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104 Department of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104 marksm@mail.med.upenn.edu.
Subba Rao Gangi SettyDepartment of Pathology and Laboratory Medicine, Department of Physiology, and Penn Vision Research Center, University of Pennsylvania, Philadelphia, PA 19104 Department of Microbiology and Cell Biology, Indian Institute of Science, Bangalore, India 560 012.
Penn Center for AIDS Research · USChildren's Hospital of Philadelphia · USCentre National pour la Recherche Scientifique et Technique (CNRST) · MASt George's, University of London · GBIndian Institute of Science Bangalore · INIn Silico Biosciences (United States) · US
Funding
UNIVERSITY OF PENNSYLVANIA CAN CTR SUPPORT GRANTP30CA016520 · NCI · UNIVERSITY OF PENNSYLVANIA · PI SHELLEY L BERGER · 1985 to 2026
$222.3MUniversity of Pennsylvania Postdoctoral Opportunities in Research and TeachingK12GM081259 · NIGMS · UNIVERSITY OF PENNSYLVANIA · PI Janis K. Burkhardt · 2007 to 2026
$20.1MScientific TransparencyP30EY001583 · NEI · UNIVERSITY OF PENNSYLVANIA · PI Qi N Cui · 1985 to 2026
$19.5MHermansky Pudlak Syndrome &melanosome formationR01EY015625 · NEI · UNIVERSITY OF PENNSYLVANIA · PI Michael S Marks · 2004 to 2026
$9.2MLamellar Body Biogenesis in Health and DiseaseR01GM108807 · NIGMS · CHILDREN'S HOSP OF PHILADELPHIA · PI GUTTENTAG, SUSAN H., MARKS, MICHAEL S · 2014 to 2017
$1.5MBLOC-1 and BLOC-2 function in melanosome maturationF32AR062476 · NIAMS · UNIVERSITY OF PENNSYLVANIA · PI DENNIS, MEGAN KATHLEEN · 2012 to 2014
$164kDBT-Wellcome Trust India Alliance 500122-Z-09-ZNCI NIH HHS P30 CA016520NEI NIH HHS P30 EY001583NEI NIH HHS R01 EY015625NIAMS NIH HHS F32 AR062476NIGMS NIH HHS K12 GM081259NIGMS NIH HHS R01 GM108807Wellcome TrustWellcome Trust 078327Wellcome Trust 500122/Z/09/Z
Abstract
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 melanosomal protein TYRP1 was largely depleted from pigment granules and underwent accelerated recycling from endosomes to the plasma membrane and to the Golgi. By live-cell imaging, recycling endosomal tubules of wild-type melanocytes made frequent and prolonged contacts with maturing melanosomes; in contrast, tubules from BLOC-2-deficient cells were shorter in length and made fewer, more transient contacts with melanosomes. These results support a model in which BLOC-2 functions to direct recycling endosomal tubular transport intermediates to maturing melanosomes and thereby promote cargo delivery and optimal pigmentation.
Indexed as
AnimalsEndocytosisEndosomesGolgi ApparatusHEK293 CellsHumansIntracellular Signaling Peptides and ProteinsMelanocytesMelanosomesMembrane GlycoproteinsMice, Inbred C57BLMice, TransgenicOxidoreductasesProtein TransportSkin PigmentationVesicular Transport ProteinsHps6 protein, mouseIntracellular Signaling Peptides and ProteinsMembrane GlycoproteinsOxidoreductasesTyrp1 protein, mouseVesicular Transport Proteins
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