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Lysosomal Targeting of Cystinosin Requires AP ‐3
Author(s) -
Andrzejewska Zuzanna,
Névo Nathalie,
Thomas Lucie,
Bailleux Anne,
Chauvet Véronique,
Benmerah Alexandre,
Antignac Corinne
Publication year - 2015
Publication title -
traffic
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 2.677
H-Index - 130
eISSN - 1600-0854
pISSN - 1398-9219
DOI - 10.1111/tra.12277
Subject(s) - microbiology and biotechnology , biology , endocytosis , endosome , signal transducing adaptor protein , cystinosis , clathrin , biochemistry , intracellular , signal transduction , cell , cystine , cysteine , enzyme
Cystinosin, a seven transmembrane ( TM ) lysosomal cystine transporter defective in the autosomal recessive lysosomal storage disorder, has been shown to be targeted to lysosomes via two sorting signals: a tyrosine‐based motif in its C‐terminal ( GYDQL ) and a non‐classical motif in its fifth inter‐ TM loop. Here, we show that the tyrosine‐based motif specifically interacts with AP ‐3 complex and functions as a ‘strong’ AP ‐3 motif responsible for direct intracellular targeting of cystinosin and the CD63 ‐cystinosin chimera ( CD63‐GYDQL ) to late endosomes/lysosomes.