{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,21]],"date-time":"2026-09-21T11:42:48Z","timestamp":1789990968301,"version":"4.0.1"},"reference-count":43,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2006,1,9]],"date-time":"2006-01-09T00:00:00Z","timestamp":1136764800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Traffic"],"published-print":{"date-parts":[[2006,3]]},"abstract":"<jats:p><jats:bold>Mutations in the mucolipin\u20101 gene have been linked to mucolipidosis type IV, a lysosomal storage disorder characterized by severe neurological and ophthalmologic abnormalities. Mucolipin\u20101 is a membrane protein containing six putative transmembrane domains with both its N\u2010 and C\u2010termini localized facing the cytosol. To gain information on the sorting motifs that mediate the trafficking of this protein to lysosomes, we have generated chimeras in which the N\u2010 and C\u2010 terminal tail portions of mucolipin\u20101 were fused to a reporter gene. In this article, we report the identification of two separate di\u2010leucine\u2010type motifs that co\u2010operate to regulate the transport of mucolipin\u20101 to lysosomes. One di\u2010leucine motif is positioned at the N\u2010terminal cytosolic tail and mediates direct transport to lysosomes, whereas the other di\u2010leucine motif is found at the C\u2010terminal tail and functions as an adaptor protein 2\u2010dependent internalization motif. We have also found that the C\u2010terminal tail of mucolipin\u20101 is palmitoylated and that this modification might regulate the efficiency of endocytosis. Finally, the mutagenesis of both di\u2010leucine motifs abrogated lysosomal accumulation and resulted in cell\u2010surface redistribution of mucolipin\u20101. Taken together, these results reveal novel information regarding the motifs that regulate mucolipin\u20101 trafficking and suggest a role for palmitoylation in protein sorting.<\/jats:bold><\/jats:p>","DOI":"10.1111\/j.1600-0854.2006.00387.x","type":"journal-article","created":{"date-parts":[[2006,1,9]],"date-time":"2006-01-09T14:24:57Z","timestamp":1136816697000},"page":"337-353","source":"Crossref","is-referenced-by-count":155,"title":["Two Di\u2010Leucine Motifs Regulate Trafficking of Mucolipin\u20101 to 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