Not just pretty eyes: Drosophila eye-colour mutations and lysosomal delivery

Lloyd, Vett K.; Ramaswami, Mani; Krämer, Helmut

doi:10.1016/s0962-8924(98)01270-7

Cited by 155 publications

(151 citation statements)

References 21 publications

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“…Interestingly, besides Tcbw, the X appears to have only two other orthologs to Drosophila eye-color genes (Table 1): cardinal, an enzyme of the ommochrome pathway (Ferre et al 1986;Tearle 1991), and ruby, which encodes a lysosomal protein important in the formation of pigment granules (Mullins et al 2000), in which the final pigments of both pathways are created and stored (Ferre et al 1986;Lloyd et al 1998). Because of ruby's role in formation of lysosome-related organelles, it seems likely that this gene, and its role in pigment granule formation, would be conserved in Tribolium.…”

Section: Analysis Of Mutant Linesmentioning

confidence: 99%

The ABCs of Eye Color inTribolium castaneum: Orthologs of theDrosophila white,scarlet, andbrownGenes

et al. 2015

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In Drosophila melanogaster, each of the three paralogous ABC transporters, White, Scarlet and Brown, is required for normal pigmentation of the compound eye. We have cloned the three orthologous genes from the beetle Tribolium castaneum. Conceptual translations of Tribolium white (Tcw), scarlet (Tcst), and brown (Tcbw) are 51, 48, and 32% identical to their respective Drosophila counterparts. We have identified loss-of-eye-pigment strains that bear mutations in Tcw and Tcst: the Tcw gene in the ivory (i) strain carries a single-base transversion, which leads to an E / D amino-acid substitution in the highly conserved Walker B motif, while the Tcst gene in the pearl (p) strain has a deletion resulting in incorporation of a premature stop codon. In light of these findings, the mutant strains i and p are herein renamed white ivory (w i ) and scarlet pearl (st p ), respectively. In addition, RNA inhibition of Tcw and Tcst recapitulates the mutant phenotypes, confirming the roles of these genes in normal eye pigmentation, while RNA interference of Tcbw provides further evidence that it has no role in eye pigmentation in Tribolium. We also consider the evolutionary implications of our findings.

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Section: Analysis Of Mutant Linesmentioning

confidence: 99%

The ABCs of Eye Color inTribolium castaneum: Orthologs of theDrosophila white,scarlet, andbrownGenes

et al. 2015

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“…Transport proteins that carry degradative enzymes to the lysosome must then be recycled back to the Golgi. Interestingly, genes in the direct AP-3 pathway have been shown to be required for the formation of pigment granules, and mutations in Drosophila and mouse AP-3 subunits lead to eye and coat color phenotypes, respectively (Lloyd et al 1998;Odorizzi et al 1998b). Not surprisingly, this vast array of membrane trafficking events requires a complex network of players to get the right cargo to the right place.…”

Section: Endosomal Sorting and Late Endosome Formationmentioning

confidence: 99%

When cell biology meets development: endocytic regulation of signaling pathways

2002

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“…For example, the HOPS complex proteins (Vps11p, Vps16p, Vps18p, and Vps33p) regulate membrane fusion events necessary for lysosomal delivery within yeast (Rieder and Emr, 1997;Peterson and Emr, 2001), Drosophila melanogaster (Sevrioukov et al, 1999;Sriram et al, 2003), and mammalian (Poupon et al, 2003;Richardson et al, 2004) endosomal systems. Similarly, the proteins composing the AP-3 complex control the formation of transport vesicles for lysosomal cargo in each of these species (Odorizzi et al, 1998;Bonifacino and Traub, 2003;Luzio et al, 2003).Although the yeast vacuole has proven to be a useful model for studying basic features of lysosomes, studies in Drosophila, mammals, and Caenorhabditis elegans have identified genes necessary for the formation of lysosomes that are not conserved in yeast (Lloyd et al, 1998;Spritz, 1999;Marks and Seabra, 2001;Piper and Luzio, 2004;Treusch et al, 2004). For example, the Hook family of proteins are present in Drosophila (Kramer and Phistry, 1996), C. elegans (Malone et al, 2003), and mammals but not in yeast (Walenta et al, 2001).…”

mentioning

confidence: 99%

“…In addition, animal cells can contain specialized lysosome-related organelles that are not found in yeast (Dell'Angelica et al, 2000b). Examples include melanosomes in vertebrate melanocytes and pigment granules in Drosophila retinal pigment cells (Lloyd et al, 1998;Marks and Seabra, 2001;Raposo and Marks, 2002 diseases are associated with defects in the formation or function of these specialized lysosomes, such as HermanskyPudlak syndrome (HPS), Chediak-Higashi syndrome, and Griscelli syndrome (Stinchcombe et al, 2004). HPS in mammals is a genetic disorder characterized by partial albinism and prolonged bleeding resulting from defects in melanosome and platelet-dense granule formation, respectively (Huizing et al, 2002;Li et al, 2004).…”

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confidence: 99%

Genetic Analysis of Lysosomal Trafficking inCaenorhabditis elegans

Hermann

Schroeder

Hieb

et al. 2005

MBoC

263

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The intestinal cells of Caenorhabditis elegans embryos contain prominent, birefringent gut granules that we show are lysosome-related organelles. Gut granules are labeled by lysosomal markers, and their formation is disrupted in embryos depleted of AP-3 subunits, VPS-16, and VPS-41. We define a class of gut granule loss (glo) mutants that are defective in gut granule biogenesis. We show that the glo-1 gene encodes a predicted Rab GTPase that localizes to lysosome-related gut granules in the intestine and that glo-4 encodes a possible GLO-1 guanine nucleotide exchange factor. These and other glo genes are homologous to genes implicated in the biogenesis of specialized, lysosome-related organelles such as melanosomes in mammals and pigment granules in Drosophila. The glo mutants thus provide a simple model system for the analysis of lysosome-related organelle biogenesis in animal cells. INTRODUCTIONLysosomes are ubiquitous membrane-bound organelles that function as major degradative sites within eukaryotic cells (Tappel, 1969). Lysosomes contain an assortment of aciddependent hydrolases that function in the breakdown of proteins, lipids, nucleic acids, and oligosaccharides. Lysosomes receive exogenous material through the endocytic pathway and are characterized as being the terminal compartment of the endocytic pathway. Lysosomes also receive material via the secretory pathway and directly from the cytoplasm (Kornfeld and Mellman, 1989;Mullins and Bonifacino, 2001;Luzio et al., 2003). Lysosomes function in diverse and important cellular processes including cell surface receptor turnover, destruction of pathogens, antigen processing, digestion, starvation responses, tissue remodeling, ion storage, autophagy, and plasma membrane repair.The yeast vacuole shares several characteristics with the lysosomes of higher animals. Genetic screens have led to the identification of Ͼ150 genes necessary for the transport and sorting of newly synthesized proteins to the yeast vacuole (Jones, 1977;Bankaitis et al., 1986;Rothman and Stevens, 1986;Bonangelino et al., 2002). These genes control two pathways of Golgi-to-vacuole transport, the carboxypeptidase Y (CPY) and alkaline phosphatase (ALP) sorting pathways (Burd et al., 1998;Conibear and Stevens, 1998;Mullins and Bonifacino, 2001). Proteins trafficked via the CPY pathway transit an endosomal prevacuolar compartment en route to the vacuole. The ALP pathway mediates transport to the vacuole independent of the prevacuolar compartment.Many of the genes involved in transport to the yeast vacuole have homologues in higher animals (Lemmon and Traub, 2000;Mullins and Bonifacino, 2001;Bonangelino et al., 2002). For example, the HOPS complex proteins (Vps11p, Vps16p, Vps18p, and Vps33p) regulate membrane fusion events necessary for lysosomal delivery within yeast (Rieder and Emr, 1997;Peterson and Emr, 2001), Drosophila melanogaster (Sevrioukov et al., 1999;Sriram et al., 2003), and mammalian (Poupon et al., 2003;Richardson et al., 2004) endosomal systems. Similarly, the proteins compos...

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Not just pretty eyes: Drosophila eye-colour mutations and lysosomal delivery

Cited by 155 publications

References 21 publications

The ABCs of Eye Color inTribolium castaneum: Orthologs of theDrosophila white,scarlet, andbrownGenes

The ABCs of Eye Color inTribolium castaneum: Orthologs of theDrosophila white,scarlet, andbrownGenes

When cell biology meets development: endocytic regulation of signaling pathways

Genetic Analysis of Lysosomal Trafficking inCaenorhabditis elegans

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