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BLOC-2 targets recycling endosomal tubules to melanosomes for cargo delivery.

Dennis, MK; Mantegazza, AR; Snir, OL; Tenza, D; Acosta-Ruiz, A; Delevoye, C; Zorger, R; Sitaram, A; de Jesus-Rojas, W; Ravichandran, K; et al. Dennis, MK; Mantegazza, AR; Snir, OL; Tenza, D; Acosta-Ruiz, A; Delevoye, C; Zorger, R; Sitaram, A; de Jesus-Rojas, W; Ravichandran, K; Rux, J; Sviderskaya, EV; Bennett, DC; Raposo, G; Marks, MS; Setty, SRG (2015) BLOC-2 targets recycling endosomal tubules to melanosomes for cargo delivery. Journal of Cell Biology, 209 (4). pp. 563-577. ISSN 1540-8140 https://doi.org/10.1083/jcb.201410026
SGUL Authors: Bennett, Dorothy Catherine Sviderskaya, Elena Vladimirovna

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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.

Item Type: Article
Additional Information: © 2015 Dennis et al. This article is distributed under the terms of an Attribution–Noncommercial–Share Alike–No Mirror Sites license for the first six months after the publication date (see http://www.rupress.org/terms). After six months it is available under a Creative Commons License (Attribution–Noncommercial–Share Alike 3.0 Unported license, as described at http://creativecommons.org/licenses/by-nc-sa/3.0/).
Keywords: Animals, Endocytosis, Endosomes, Golgi Apparatus, HEK293 Cells, Humans, Melanocytes, Melanosomes, Membrane Glycoproteins, Mice, Inbred C57BL, Mice, Transgenic, Oxidoreductases, Protein Transport, Skin Pigmentation, Vesicular Transport Proteins, Melanosomes, Endosomes, Golgi Apparatus, Melanocytes, Animals, Mice, Inbred C57BL, Mice, Transgenic, Humans, Oxidoreductases, Membrane Glycoproteins, Vesicular Transport Proteins, Skin Pigmentation, Endocytosis, Protein Transport, HEK293 Cells, Developmental Biology, 06 Biological Sciences, 11 Medical And Health Sciences
SGUL Research Institute / Research Centre: Academic Structure > Molecular and Clinical Sciences Research Institute (MCS)
Academic Structure > Molecular and Clinical Sciences Research Institute (MCS) > Cell Sciences (INCCCS)
Journal or Publication Title: Journal of Cell Biology
ISSN: 1540-8140
Language: ENG
Dates:
DateEvent
25 May 2015Published
20 April 2015Accepted
Publisher License: Creative Commons: Attribution-Noncommercial-Share Alike 3.0
Projects:
Project IDFunderFunder ID
500122/Z/09/ZWellcome Trusthttp://dx.doi.org/10.13039/100004440
R01 GM108807National Institute of General Medical Scienceshttp://dx.doi.org/10.13039/100000057
F32 AR062476National Institute of Arthritis and Musculoskeletal and Skin Diseaseshttp://dx.doi.org/10.13039/100000069
R01 EY015625National Eye Institutehttp://dx.doi.org/10.13039/100000053
P30 EY001583National Eye Institutehttp://dx.doi.org/10.13039/100000053
P30 CA016520National Cancer Institutehttp://dx.doi.org/10.13039/100000054
K12 GM081259National Institute of General Medical Scienceshttp://dx.doi.org/10.13039/100000057
078327Wellcome Trusthttp://dx.doi.org/10.13039/100004440
PubMed ID: 26008744
Web of Science ID: WOS:000355643900013
Go to PubMed abstract
URI: https://openaccess.sgul.ac.uk/id/eprint/108429
Publisher's version: https://doi.org/10.1083/jcb.201410026

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