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PMID:15563610

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Citation

Hu, J and Barr, MM (2005) ATP-2 interacts with the PLAT domain of LOV-1 and is involved in Caenorhabditis elegans polycystin signaling. Mol. Biol. Cell 16:458-69

Abstract

Caenorhabditis elegans is a powerful model to study the molecular basis of autosomal dominant polycystic kidney disease (ADPKD). ADPKD is caused by mutations in the polycystic kidney disease (PKD)1 or PKD2 gene, encoding polycystin (PC)-1 or PC-2, respectively. The C. elegans polycystins LOV-1 and PKD-2 are required for male mating behaviors and are localized to sensory cilia. The function of the evolutionarily conserved polycystin/lipoxygenase/alpha-toxin (PLAT) domain found in all PC-1 family members remains an enigma. Here, we report that ATP-2, the beta subunit of the ATP synthase, physically associates with the LOV-1 PLAT domain and that this interaction is evolutionarily conserved. In addition to the expected mitochondria localization, ATP-2 and other ATP synthase components colocalize with LOV-1 and PKD-2 in cilia. Disrupting the function of the ATP synthase or overexpression of atp-2 results in a male mating behavior defect. We further show that atp-2, lov-1, and pkd-2 act in the same molecular pathway. We propose that the ciliary localized ATP synthase may play a previously unsuspected role in polycystin signaling.

Links

PubMed PMC545878 Online version:10.1091/mbc.E04-09-0851

Keywords

Animals; Animals, Genetically Modified; Caenorhabditis elegans/genetics; Caenorhabditis elegans/metabolism; Caenorhabditis elegans Proteins/chemistry; Caenorhabditis elegans Proteins/genetics; Caenorhabditis elegans Proteins/metabolism; Cation Transport Proteins; Genes, Helminth/genetics; Humans; Male; Membrane Proteins/metabolism; Mitochondrial Proton-Translocating ATPases/chemistry; Mitochondrial Proton-Translocating ATPases/metabolism; Nerve Tissue Proteins/genetics; Nerve Tissue Proteins/metabolism; Neurons, Afferent/metabolism; Polycystic Kidney, Autosomal Dominant/genetics; Polycystic Kidney, Autosomal Dominant/metabolism; Protein Binding; Protein Structure, Tertiary; Protein Subunits/chemistry; Protein Subunits/metabolism; Proteins/metabolism; RNA Interference; Sexual Behavior, Animal; Signal Transduction; TRPP Cation Channels; Two-Hybrid System Techniques; Zebrafish Proteins/genetics; Zebrafish Proteins/metabolism

Significance

Annotations

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

CAEEL:ATPB

enables

GO:0005515: protein binding

ECO:0000353: physical interaction evidence used in manual assertion

UniProtKB:Q09624

F

Seeded From UniProt

complete

CAEEL:ATPB

enables

GO:0019904: protein domain specific binding

ECO:0000353: physical interaction evidence used in manual assertion

WB:WBGene00003058

F

Seeded From UniProt

complete

CAEEL:ATPB

enables

GO:0019904: protein domain specific binding

ECO:0000353: physical interaction evidence used in manual assertion

UniProtKB:P98161

F

Seeded From UniProt

complete

CAEEL:ATPB

located_in

GO:0097730: non-motile cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:ATPB

involved_in

GO:0007617: mating behavior

ECO:0000315: mutant phenotype evidence used in manual assertion

WB:WBRNAi00063768
WB:WBRNAi00063778
WB:WBRNAi00063779

P

Seeded From UniProt

complete

CAEEL:ATPB

located_in

GO:0005739: mitochondrion

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:ATPB

enables

GO:0005515: protein binding

ECO:0000353: physical interaction evidence used in manual assertion

UniProtKB:Q09624

F

Seeded From UniProt

complete

CAEEL:ATPB

located_in

GO:0005929: cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

HUMAN:PKD1

enables

GO:0005515: protein binding

ECO:0000353: physical interaction evidence used in manual assertion

UniProtKB:P46561

F

Seeded From UniProt

complete

CAEEL:PKD1

enables

GO:0005515: protein binding

ECO:0000353: physical interaction evidence used in manual assertion

UniProtKB:P46561

F

Seeded From UniProt

complete

CAEEL:PKD1

enables

GO:0019904: protein domain specific binding

ECO:0000353: physical interaction evidence used in manual assertion

WB:WBGene00000229

F

Seeded From UniProt

complete

CAEEL:PKD1

involved_in

GO:0007617: mating behavior

ECO:0000315: mutant phenotype evidence used in manual assertion

WB:WBVar00249016

P

Seeded From UniProt

complete

CAEEL:PKD1

enables

GO:0005515: protein binding

ECO:0000353: physical interaction evidence used in manual assertion

UniProtKB:P46561

F

Seeded From UniProt

complete

CAEEL:PKD1

located_in

GO:0005929: cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:PKD1

involved_in

GO:0060179: male mating behavior

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

CAEEL:PKD1

involved_in

GO:0034608: vulval location

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

CAEEL:PKD1

involved_in

GO:0034606: response to hermaphrodite contact

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

CAEEL:ATPL2

located_in

GO:0097730: non-motile cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:ATPL2

located_in

GO:0005739: mitochondrion

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:Q20053

located_in

GO:0097730: non-motile cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:Q20053

located_in

GO:0005739: mitochondrion

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:PKD2

located_in

GO:0097730: non-motile cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:PKD2

located_in

GO:0030424: axon

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:PKD2

located_in

GO:0030425: dendrite

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

CAEEL:PKD2

located_in

GO:0043025: neuronal cell body

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

See also

References

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