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PMID:20223935

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Citation

Miller, RM and Portman, DS A latent capacity of the C. elegans polycystins to disrupt sensory transduction is repressed by the single-pass ciliary membrane protein CWP-5. Dis Model Mech 3:441-50

Abstract

Autosomal dominant polycystic kidney disease (ADPKD) results from loss-of-function mutations in PKD1 or PKD2. The products of these genes, the polycystins PC-1 and PC-2, form a transmembrane channel that is necessary for flow sensing by renal cilia. In C. elegans, the polycystin orthologs LOV-1 and PKD-2 function in sensory neurons that mediate male mating behavior. Here, we report that the novel single-pass membrane protein CWP-5 is necessary for polycystin signaling during the response step of mating behavior. As with the polycystins, CWP-5 localizes to neuronal cilia; this localization requires LOV-1. The response defect of cwp-5 mutants does not appear to result from disruption of ciliogenesis or polycystin localization. Instead, genetic and behavioral analyses indicate that CWP-5 represses a previously undescribed antagonistic effect of the polycystins on sensory function. Although cwp-5 does not have a primary-sequence ortholog in vertebrates, it has intriguing parallels with the autosomal recessive PKD gene FPC (also known as PKHD1). Together, this study identifies a new component of C. elegans polycystin signaling, demonstrates that the polycystins have a latent capacity to hinder sensory transduction, and suggests that aberrant functions of the polycystins could contribute to the pathogenesis of PKD.

Links

PubMed PMC2898535 Online version:10.1242/dmm.002816

Keywords

Animals; Behavior, Animal; Caenorhabditis elegans/cytology; Caenorhabditis elegans/genetics; Caenorhabditis elegans/metabolism; Caenorhabditis elegans Proteins/genetics; Caenorhabditis elegans Proteins/metabolism; Cilia/metabolism; Gene Expression Regulation; Genes, Helminth; Intracellular Membranes/metabolism; Male; Membrane Proteins/metabolism; Models, Genetic; Mutation/genetics; Protein Transport; Sensory Receptor Cells/metabolism; Sex Characteristics; Signal Transduction; TRPP Cation Channels/metabolism

Significance

Annotations

Gene product Qualifier GO Term Evidence Code with/from Aspect Extension Notes Status

CAEEL:B6VQ87

involved_in

GO:0023041: neuronal signal transduction

ECO:0000316: genetic interaction evidence used in manual assertion

WB:WBGene00004035

P

Seeded From UniProt

complete

CAEEL:B6VQ87

involved_in

GO:0023041: neuronal signal transduction

ECO:0000316: genetic interaction evidence used in manual assertion

WB:WBGene00003058

P

Seeded From UniProt

complete

CAEEL:B6VQ87

located_in

GO:0097730: non-motile cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:B6VQ87

located_in

GO:0030425: dendrite

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:B6VQ87

located_in

GO:0044297: cell body

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:B6VQ87

involved_in

GO:0034606: response to hermaphrodite contact

ECO:0000315: mutant phenotype evidence used in manual assertion

WB:WBVar00250856

P

Seeded From UniProt

complete

CAEEL:B6VQ88

involved_in

GO:0023041: neuronal signal transduction

ECO:0000316: genetic interaction evidence used in manual assertion

WB:WBGene00004035

P

Seeded From UniProt

complete

CAEEL:B6VQ88

involved_in

GO:0023041: neuronal signal transduction

ECO:0000316: genetic interaction evidence used in manual assertion

WB:WBGene00003058

P

Seeded From UniProt

complete

CAEEL:B6VQ88

located_in

GO:0030425: dendrite

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:B6VQ88

located_in

GO:0005929: cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:B6VQ88

located_in

GO:0044297: cell body

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:B6VQ88

involved_in

GO:0034606: response to hermaphrodite contact

ECO:0000315: mutant phenotype evidence used in manual assertion

WB:WBVar00250856

P

Seeded From UniProt

complete


See also

References

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