CUPpling calcium to lysosomal biogenesis.

Transport from late endosomes to lysosomes results in the formation of an endosome-lysosome hybrid organelle from which late endosomes and lysosomes must be re-formed. Recent studies indicate that the transient receptor potential (TRP)-related channel mucolipin-1 (the loss of which causes mucolipidosis type IV) and its Caenorhabditis elegans orthologue CUP-5 might control the process of lysosome re-formation by regulating calcium flux.

[1]  I. Greenwald,et al.  Regulation of endocytosis by CUP-5, the Caenorhabditis elegans mucolipin-1 homolog , 2001, Nature Genetics.

[2]  G. Bach.  Mucolipidosis type IV. , 2001, Molecular genetics and metabolism.

[3]  R. Murphy,et al.  Endosome maturation: insights from somatic cell genetics and cell-free analysis. , 1993, Biochemical Society transactions.

[4]  B. Minke,et al.  TRP channel proteins and signal transduction. , 2002, Physiological reviews.

[5]  J. Luzio,et al.  Fusion of Lysosomes with Late Endosomes Produces a Hybrid Organelle of Intermediate Density and Is NSF Dependent , 1998, The Journal of cell biology.

[6]  Gong Chen,et al.  Formation of an Endophilin-Ca2+ Channel Complex Is Critical for Clathrin-Mediated Synaptic Vesicle Endocytosis , 2003, Cell.

[7]  W. Wickner,et al.  Trans-SNARE interactions elicit Ca2+ efflux from the yeast vacuole lumen , 2004, The Journal of cell biology.

[8]  I. Mills,et al.  Clathrin adaptor epsinR is required for retrograde sorting on early endosomal membranes. , 2004, Developmental cell.

[9]  R. Pagano,et al.  Abnormal transport along the lysosomal pathway in mucolipidosis, type IV disease. , 1998, Proceedings of the National Academy of Sciences of the United States of America.

[10]  H. Geuze,et al.  Transport from late endosomes to lysosomes, but not sorting of integral membrane proteins in endosomes, depends on the vacuolar proton pump , 1995, The Journal of cell biology.

[11]  C. Kung,et al.  The transient receptor potential channel on the yeast vacuole is mechanosensitive , 2003, Proceedings of the National Academy of Sciences of the United States of America.

[12]  M. Mann,et al.  Control of the terminal step of intracellular membrane fusion by protein phosphatase 1. , 1999, Science.

[13]  G. Baldwin,et al.  Calcium Binds Dynamin I and Inhibits Its GTPase Activity , 1996, Journal of neurochemistry.

[14]  S. Slaugenhaupt,et al.  Caenorhabditis elegans functional orthologue of human protein h-mucolipin-1 is required for lysosome biogenesis. , 2004, Proceedings of the National Academy of Sciences of the United States of America.

[15]  D. Hirsh,et al.  RME-8, a conserved J-domain protein, is required for endocytosis in Caenorhabditis elegans. , 2001, Molecular biology of the cell.

[16]  L. Wan,et al.  PACS-1 Defines a Novel Gene Family of Cytosolic Sorting Proteins Required for trans-Golgi Network Localization , 1998, Cell.

[17]  C. Futter,et al.  Multivesicular endosomes containing internalized EGF-EGF receptor complexes mature and then fuse directly with lysosomes , 1996, The Journal of cell biology.

[18]  A. Mayer,et al.  Ca2+/calmodulin signals the completion of docking and triggers a late step of vacuole fusion , 1998, Nature.

[19]  D. Lancet,et al.  Mucolipidosis type IV: Novel MCOLN1 mutations in Jewish and non‐Jewish patients and the frequency of the disease in the Ashkenazi Jewish population , 2001, Human mutation.

[20]  M. Desjardins,et al.  Remodeling of endosomes during lysosome biogenesis involves `kiss and run' fusion events regulated by rab5 , 2003, Journal of Cell Science.

[21]  Barbara J. Reaves,et al.  Dense core lysosomes can fuse with late endosomes and are re-formed from the resultant hybrid organelles. , 1997, Journal of cell science.

[22]  S. Pfeffer,et al.  Membrane transport: Retromer to the rescue , 2001, Current Biology.

[23]  J. Meldolesi,et al.  Synaptic vesicles: is kissing a matter of competence? , 2001, Trends in cell biology.

[24]  M. Stamnes,et al.  A Role for Calcium in Stabilizing Transport Vesicle Coats* , 2001, The Journal of Biological Chemistry.

[25]  P. Robinson,et al.  The dephosphins: dephosphorylation by calcineurin triggers synaptic vesicle endocytosis , 2001, Trends in Neurosciences.

[26]  W. H. Goldmann,et al.  Molecular pathophysiology of mucolipidosis type IV: pH dysregulation of the mucolipin-1 cation channel. , 2004, Human molecular genetics.

[27]  Erika Hartwieg,et al.  The Caenorhabditis elegans mucolipin-like gene cup-5 is essential for viability and regulates lysosomes in multiple cell types , 2002, Proceedings of the National Academy of Sciences of the United States of America.

[28]  Jesse T. Myers,et al.  pH-dependent regulation of lysosomal calcium in macrophages. , 2002, Journal of cell science.

[29]  B. Storrie,et al.  The biogenesis of lysosomes: Is it a kiss and run, continuous fusion and fission process? , 1996, BioEssays : news and reviews in molecular, cellular and developmental biology.

[30]  J. Luzio,et al.  Membrane dynamics and the biogenesis of lysosomes (Review) , 2003 .

[31]  J. Luzio,et al.  The Role of Intraorganellar Ca2+In Late Endosome–Lysosome Heterotypic Fusion and in the Reformation of Lysosomes from Hybrid Organelles , 2000, The Journal of cell biology.

[32]  E. Brown,et al.  Identification and characterization of the single channel function of human mucolipin‐1 implicated in mucolipidosis type IV, a disorder affecting the lysosomal pathway , 2002, FEBS letters.

[33]  R. Schiffmann,et al.  The neurogenetics of mucolipidosis type IV , 2002, Neurology.