Lowe syndrome protein OCRL1 interacts with clathrin and regulates protein trafficking between endosomes and the trans-Golgi network

被引:149
作者
Choudhury, R
Diao, AP
Zhang, F
Eisenberg, E
Saint-Pol, A
Williams, C
Konstantakopoulos, A
Lucocq, J
Johannes, L
Rabouille, C
Greene, LE
Lowe, M [1 ]
机构
[1] Univ Manchester, Fac Life Sci, Manchester M13 9PT, Lancs, England
[2] NHLBI, Cell Biol Lab, NIH, Bethesda, MD 20892 USA
[3] Inst Curie, Dept Cellular Compartmentalizat & Dynam, Traffic & Signaling Lab, CNRS,Unite Mixte Rech 144, F-75248 Paris, France
[4] Univ Utrecht, Ctr Med, Dept Cell Biol, AZU, NL-3584 CX Utrecht, Netherlands
[5] Univ Dundee, Sch Life Sci, Dundee DD1 5EH, Scotland
关键词
D O I
10.1091/mbc.E05-02-0120
中图分类号
Q2 [细胞生物学];
学科分类号
071009 ; 090102 ;
摘要
Oculocerebrorenal syndrome of Lowe is caused by mutation of OCRL1, a phosphatidylinositol 4,5-bisphosphate 5-phosphatase localized at the Golgi apparatus. The cellular role of OCRL1 is unknown, and consequently the mechanism by which loss of OCRL1 function leads to disease is ill defined. Here, we show that OCRL1 is associated with clathrin-coated transport intermediates operating between the trans-Golgi network (TGN) and endosomes. OCRL1 interacts directly with clathrin heavy chain and promotes clathrin assembly in vitro. Interaction with clathrin is not, however, required for membrane association of OCRL1. Overexpression of OCRL1 results in redistribution of clathrin and the cation-independent mannose 6-phosphate receptor (CI-MPR) to enlarged endosomal structures that are defective in retrograde trafficking to the TGN. Depletion of cellular OCRL1 also causes partial redistribution of a CI-MPR reporter to early endosomes. These findings suggest a role for OCRL1 in clathrin-mediated trafficking of proteins from endosomes to the TGN and that defects in this pathway might contribute to the Lowe syndrome phenotype.
引用
收藏
页码:3467 / 3479
页数:13
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