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These pigmentation defects indicate a key but yet unexplored physiological relevance of this complex in the biogenesis of melanosomes. Here we show that PIKfyve activity regulates formation of amyloid matrix composed of PMEL protein within early endosomes, called stage I melanosomes. PIKfyve activity controls the membrane remodeling of stage I melanosomes that increases PMEL abundance and impairs its sorting and processing. PIKfyve activity also affects stage I melanosome kiss-and-run interactions with lysosomes that is required for PMEL amyloidogenesis and establishment of melanosome identity. Mechanistically, PIKfyve activity promotes the formation and membrane tubules from stage I melanosomes and their release by modulating endosomal actin branching. Together our data indicate that PIKfyve activity is a key regulator of the melanosomal import-export machinery that fine tunes the formation of functional amyloid fibrils in melanosomes and the maintenance of melanosome identity.<\/jats:p>","DOI":"10.1242\/jcs.229500","type":"journal-article","created":{"date-parts":[[2019,2,1]],"date-time":"2019-02-01T15:27:08Z","timestamp":1549034828000},"source":"Crossref","is-referenced-by-count":30,"title":["PIKfyve complex regulates early melanosome homeostasis required for physiological amyloid formation"],"prefix":"10.1242","author":[{"given":"Christin","family":"Bissig","sequence":"first","affiliation":[{"name":"Structure and Membrane Compartments, Institut Curie, Paris Sciences & Lettres Research University, Centre National de la Recherche Scientifique, UMR144, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pauline","family":"Crois\u00e9","sequence":"additional","affiliation":[{"name":"IPNP, Institute of Psychiatry and Neuroscience of Paris, Hopital Saint-Anne, Universit\u00e9 Paris Descartes, INSERM U894, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xavier","family":"Heiligenstein","sequence":"additional","affiliation":[{"name":"Structure and Membrane Compartments, Institut Curie, Paris Sciences & Lettres Research University, Centre National de la Recherche Scientifique, UMR144, Paris, France"},{"name":"Cell and Tissue Imaging Facility, Institut Curie, Paris Sciences & Lettres Research University, Centre National de la Recherche Scientifique, UMR144, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ilse","family":"Hurbain","sequence":"additional","affiliation":[{"name":"Structure and Membrane Compartments, Institut Curie, Paris Sciences & Lettres Research University, Centre National de la Recherche Scientifique, UMR144, Paris, France"},{"name":"Cell and Tissue Imaging Facility, Institut Curie, Paris Sciences & Lettres Research University, Centre National de la Recherche Scientifique, UMR144, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guy M.","family":"Lenk","sequence":"additional","affiliation":[{"name":"Department of Human Genetics, University of Michigan, Ann Arbor, Michigan, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emily","family":"Kaufman","sequence":"additional","affiliation":[{"name":"Life Science Institute, University of Michigan, Ann Arbor, Michigan, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ragna","family":"Sannerud","sequence":"additional","affiliation":[{"name":"VIB Center for Brain & Disease Research, Leuven, Belgium"},{"name":"KU Leuven, Department of Neurosciences, Leuven, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wim","family":"Annaert","sequence":"additional","affiliation":[{"name":"VIB Center for Brain & Disease Research, Leuven, Belgium"},{"name":"KU Leuven, Department of Neurosciences, Leuven, Belgium"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Miriam H.","family":"Meisler","sequence":"additional","affiliation":[{"name":"Department of Human Genetics, University of Michigan, Ann Arbor, Michigan, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lois S.","family":"Weisman","sequence":"additional","affiliation":[{"name":"Life Science Institute, University of Michigan, Ann Arbor, Michigan, USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Gra\u00e7a","family":"Raposo","sequence":"additional","affiliation":[{"name":"Structure and Membrane Compartments, Institut Curie, Paris Sciences & Lettres Research University, Centre National de la Recherche Scientifique, UMR144, Paris, France"},{"name":"Cell and Tissue Imaging Facility, Institut Curie, Paris Sciences & Lettres Research University, Centre National de la Recherche Scientifique, UMR144, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guillaume","family":"van Niel","sequence":"additional","affiliation":[{"name":"Structure and Membrane Compartments, Institut Curie, Paris Sciences & Lettres Research University, Centre National de la Recherche Scientifique, UMR144, Paris, France"},{"name":"IPNP, Institute of Psychiatry and Neuroscience of Paris, Hopital Saint-Anne, Universit\u00e9 Paris Descartes, INSERM U894, Paris, France"},{"name":"Cell and Tissue Imaging Facility, Institut Curie, Paris Sciences & Lettres Research University, Centre National de la Recherche Scientifique, UMR144, Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"237","published-online":{"date-parts":[[2019,1,1]]},"reference":[{"key":"2021042422431862600_JCS229500C1","doi-asserted-by":"publisher","first-page":"158104","DOI":"10.1103\/PhysRevLett.93.158104","article-title":"Fission of a multiphase membrane tube","volume":"93","author":"Allain","year":"2004","journal-title":"Phys. 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