Abstract：
:Autosomal dominant polycystic kidney disease (ADPKD) is the most common monogenic human disease, with around 12.5 million people affected worldwide. ADPKD results from mutations in either PKD1 or PKD2, which encode the atypical G-protein coupled receptor polycystin-1 (PC1) and the transient receptor potential channel polycystin-2 (PC2), respectively. Although altered intracellular trafficking of PC1 and PC2 is an underlying feature of ADPKD, the mechanisms which govern vesicular transport of the polycystins through the biosynthetic and endosomal membrane networks remain to be fully elucidated. Here, we describe an interaction between PC2 and retromer, a master controller for the sorting of integral membrane proteins through the endo-lysosomal network. We show that association of PC2 with retromer occurs via a region in the PC2 cytoplasmic amino-terminal domain, independently of the retromer-binding Wiskott-Aldrich syndrome and scar homologue (WASH) complex. Based on observations that retromer preferentially interacts with a trafficking population of PC2, and that ciliary levels of PC1 are reduced upon mutation of key residues required for retromer association in PC2, our data are consistent with the identification of PC2 as a retromer cargo protein.This article has an associated First Person interview with the first author of the paper.
journal_name
J Cell Scijournal_title
Journal of cell scienceauthors
Tilley FC,Gallon M,Luo C,Danson CM,Zhou J,Cullen PJdoi
10.1242/jcs.211342subject
Has Abstractpub_date
2018-06-06 00:00:00issue
11eissn
0021-9533issn
1477-9137pii
jcs.211342journal_volume
131pub_type
杂志文章abstract：:In trypanosomes, the flagellum is rooted in the flagellar pocket, a surface micro-domain that is the sole site for endocytosis and exocytosis. By analysis of anterograde or retrograde intraflagellar transport in IFT88RNAi or IFT140RNAi mutant cells, we show that elongation of the new flagellum is not required for flag...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.035626
更新日期：2008-11-15 00:00:00
abstract：:Human procathepsin D carries two N-linked glycosylation sites at asparagine residues 70 and 199, widely separated on the surface of the folded protein. We created monoglycosylated procathepsin D molecules by site-directed mutagenesis in vitro of the individual glycosylation sites. With only two exceptions, all 12 muta...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：
更新日期：1995-05-01 00:00:00
abstract：:An electron microscopic investigation of the events associated with meiosis in Lilium has revealed a number of changes in both the organellar population and the other cytoplasmic components. Ribosome numbers decrease significantly in early prophase and are later replenished in the tetrads, a process most likely involv...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：
更新日期：1983-01-01 00:00:00
abstract：:Interactions between different cell types can induce distinct contact inhibition of locomotion (CIL) responses that are hypothesised to control population-wide behaviours during embryogenesis. However, our understanding of the signals that lead to cell-type specific repulsion and the precise capacity of heterotypic CI...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.223974
更新日期：2019-05-31 00:00:00
abstract：:In the fission yeast Schizosaccharomyces pombe, a series of diploid mutants divides at smaller cell sizes than wild type. In these smaller strains, the mean gene concentration (defined by previous authors as the DNA to protein ratio) is higher than in wild type. Such an increase in gene concentration should also incre...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：
更新日期：1979-02-01 00:00:00
abstract：:Human peripheral blood eosinophils attach to and flatten down onto antibody-coated surfaces and subsequently degranulate. An antibody-coated surface was prepared by treating a layer of agar, containing tetanus toxoid antigen and eosinophil chemotactic factor (ECF), with human anti-tetanus immunoglobin. Changes in eosi...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：
更新日期：1982-08-01 00:00:00
abstract：:Formation of crossovers between homologous chromosomes during meiosis is positively regulated by the ZMM proteins (also known as SIC proteins). DNA damage checkpoint proteins also promote efficient formation of interhomolog crossovers. Here, we examined, in budding yeast, the meiotic role of the heterotrimeric DNA dam...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.161554
更新日期：2015-04-15 00:00:00
abstract：:Members of the ADAMTS (a disintegrin and metalloproteinase with thrombospondin motifs) family of proteolytic enzymes are implicated in a variety of physiological processes, such as collagen maturation, organogenesis, angiogenesis, reproduction and inflammation. Moreover, deficiency or overexpression of certain ADAMTS ...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.005751
更新日期：2007-10-15 00:00:00
abstract：:Cannabinoid receptors (CBRs) belong to the G protein-coupled receptor superfamily, and activation of CBRs in salivary cells inhibits agonist-stimulated salivation and modifies saliva content. However, the role of different CBR subtypes in acinar cell physiology and in intracellular signalling remains unclear. Here, we...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.088930
更新日期：2012-04-15 00:00:00
abstract：:B chromosomes are centric chromosomal fragments present in thousands of eukaryotic genomes. Because most B chromosomes are non-essential, they can be lost without consequence. In order to persist, however, some B chromosomes can impose strong forms of intra-genomic conflict. An extreme case is the paternal sex ratio (...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.113423
更新日期：2012-11-01 00:00:00
abstract：:Barrier junctions prevent pathogen invasion and restrict paracellular leakage across epithelial sheets. To understand how one barrier junction, the septate junction (SJ), is regulated in vivo, we used fluorescence recovery after photobleaching (FRAP) to examine SJ protein dynamics in Drosophila. Most SJ-associated pro...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.087700
更新日期：2011-08-15 00:00:00
abstract：:The coordinated trafficking and tethering of membrane cargo within cells relies on the function of distinct cytoskeletal motors that are targeted to specific subcellular compartments through interactions with protein adaptors and phospholipids. The unique actin motor myosin VI functions at distinct steps during clathr...
journal_title：Journal of cell science
pub_type： 杂志文章,评审
doi：10.1242/jcs.095554
更新日期：2013-06-15 00:00:00
abstract：:Cell migration involves the dynamic formation and release of cell-substrate adhesions, where the exertion and detection of mechanical forces take place. Members of the calpain family of calcium-dependent proteases are believed to have a central role in these processes, possibly through the regulation of focal adhesion...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.036152
更新日期：2008-11-01 00:00:00
abstract：:The Down syndrome critical region (DSCR) on Chromosome 21 contains many genes whose duplication may lead to the major phenotypic features of Down syndrome and especially the associated mental retardation. However, the functions of DSCR genes are mostly unknown and their possible involvement in key brain developmental ...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.000703
更新日期：2007-06-01 00:00:00
abstract：:The neuromuscular junction (NMJ) is the largest, most-complex synapse in the human body. Motor neuron (MN) diseases, such as amyotrophic lateral sclerosis (ALS), specifically target MNs and the NMJs. However, little is known about the reasons for MN-selective neuronal and synaptic vulnerability in MN diseases. Here, u...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.234492
更新日期：2019-12-02 00:00:00
abstract：:The process of angiogenesis requires endothelial cells (ECs) to undergo profound changes in shape and polarity. Although this must involve remodelling of the EC cytoskeleton, little is known about this process or the proteins that control it. We used a co-culture assay of angiogenesis to examine the cytoskeleton of EC...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.091066
更新日期：2012-03-15 00:00:00
abstract：:Compartmentalization of surface membrane antigens into discrete regions or domains is a characteristic feature of differentiated cells. In mammalian spermatozoa at least 5 surface domains are known, implying the presence of barriers or boundaries within the plasma membrane. Using the technique of fluorescence recovery...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：
更新日期：1997-05-01 00:00:00
abstract：:The lepidopteran-specific P1 delta-endotoxin of Bacillus thuringiensis var. kurstaki HD-1 was activated in vitro using insect gut proteases and found to be highly specific for the lepidopteran cell line Choristoneura fumiferana CF1 among a wide range of lepidopteran and dipteran cell lines tested. The toxicity of P1 a...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：
更新日期：1986-07-01 00:00:00
abstract：:Endocytosis is a regulated physiological process by which membrane receptors and their extracellular ligands are internalized. After internalization, they enter the endosomal trafficking pathway for sorting and processing. Amphiphysins consist of a family of proteins conserved throughout evolution that are crucial ele...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.00403
更新日期：2003-05-15 00:00:00
abstract：:In previous work we used immunofluorescent staining with specific antibodies to study the distribution of five desmosomal antigens in the epithelia of different vertebrate animals. We showed that all five antigens were present in all epithelia studied in human, bovine, rat, guinea pig, chick and frog (Rana pipiens) ti...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：
更新日期：1984-03-01 00:00:00
abstract：:Proteins of the Wiskott-Aldrich syndrome protein (WASP) family function as nucleation-promoting factors for the ubiquitously expressed Arp2/3 complex, which drives the generation of branched actin filaments. Arp2/3-generated actin regulates diverse cellular processes, including the formation of lamellipodia and filopo...
journal_title：Journal of cell science
pub_type： 杂志文章,评审
doi：10.1242/jcs.199570
更新日期：2017-07-15 00:00:00
abstract：:The notion that cell shape and spreading can regulate cell proliferation has evolved over several years, but only recently has this been linked to forces from within and upon the cell. This emerging area of mechanical signaling is proving to be wide-spread and important for all cell types. The microenvironment that su...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.067009
更新日期：2011-04-15 00:00:00
abstract：:S100 proteins have attracted great interest in recent years because of their cell- and tissue-specific expression and association with various human pathologies. Most S100 proteins are small acidic proteins with calcium-binding domains - the EF hands. It is thought that this group of proteins carry out their cellular ...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：
更新日期：2002-08-01 00:00:00
abstract：:MOR1, the Arabidopsis thaliana homologue of the Xenopus microtubule-associated protein MAP215, is required for spatial organization of the acentrosomal microtubule arrays of plant cells. To determine how loss of MOR1 function affects microtubule dynamics, we compared various parameters of microtubule dynamics in the t...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.039065
更新日期：2008-12-15 00:00:00
abstract：:During early stages of meiotic prophase I the nucleus undergoes considerable reorganization, including the clustering of telomeres, the release of contacts between chromosomes and the nuclear membrane, the reorganization of the nucleolus, and chromatin remodeling. Using a light squashing technique for the analysis of ...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.03155
更新日期：2006-09-15 00:00:00
abstract：:The formation of complex dendritic arbors is crucial for the assembly of functional networks as abnormal dendrite formation underlies several neurodevelopmental and psychiatric disorders. Many extracellular factors have been postulated as regulators of dendritic growth. Wnt proteins play a critical role in neuronal de...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.216101
更新日期：2018-07-09 00:00:00
abstract：:Vaccinia virus B1 kinase plays a key role in viral DNA replication. The homologous mammalian vaccinia-related kinases (VRKs) are also implicated in the regulation of DNA replication, although direct evidence remains elusive. Here we show that VRK1 regulates cell cycle progression in the DNA replication period by induc...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.026757
更新日期：2008-09-15 00:00:00
abstract：:We have recently shown that leukotriene D(4) (LTD(4)) increases cell survival in intestinal epithelial cells. Here we report and explore the complementary finding that LTD(4) also enhances proliferation in these cells. This proliferative response was approximately half of that induced by epidermal growth factor (EGF) ...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：
更新日期：2002-05-01 00:00:00
abstract：:Chloride intracellular channel 3 (CLIC3) drives invasiveness of pancreatic and ovarian cancer by acting in concert with Rab25 to regulate the recycling of α5β1 integrin from late endosomes to the plasma membrane. Here, we show that in two estrogen receptor (ER)-negative breast cancer cell lines, CLIC3 has little influ...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.135947
更新日期：2014-09-15 00:00:00
abstract：:In the developing Drosophila eye, the centrioles of the differentiating retinal cells are not surrounded by the microtubule-nucleating γ-tubulin, suggesting that they are unable to organize functional microtubule-organizing centers. Consistent with this idea, Cnn and Spd-2, which are involved in γ-tubulin recruitment,...
journal_title：Journal of cell science
pub_type： 杂志文章
doi：10.1242/jcs.211441
更新日期：2018-02-22 00:00:00
