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PMID:24067530

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Citation

Jerber, J, Baas, D, Soulavie, F, Chhin, B, Cortier, E, Vesque, C, Thomas, J and Durand, B (2014) The coiled-coil domain containing protein CCDC151 is required for the function of IFT-dependent motile cilia in animals. Hum. Mol. Genet. 23:563-77

Abstract

Cilia are evolutionarily conserved organelles endowed with essential physiological and developmental functions. In humans, disruption of cilia motility or signaling leads to complex pleiotropic genetic disorders called ciliopathies. Cilia motility requires the assembly of multi-subunit motile components such as dynein arms, but mechanisms underlying their assembly pathway and transport into the axoneme are still largely unknown. We identified a previously uncharacterized coiled-coil domain containing protein CCDC151, which is evolutionarily conserved in motile ciliated species and shares ancient features with the outer dynein arm-docking complex 2 of Chlamydomonas. In Drosophila, we show that CG14127/CCDC151 is associated with motile intraflagellar transport (IFT)-dependent cilia and required for geotaxis behavior of adult flies. In zebrafish, Ccdc151 is expressed in tissues with motile cilia, and morpholino-induced depletion of Ccdc151 leads to left-right asymmetry defects and kidney cysts. We demonstrate that Ccdc151 is required for proper motile function of cilia in the Kupffer's vesicle and in the pronephros by controlling dynein arm assembly, showing that Ccdc151 is a novel player in the control of IFT-dependent dynein arm assembly in animals. However, we observed that CCDC151 is also implicated in other cellular functions in vertebrates. In zebrafish, ccdc151 is involved in proper orientation of cell divisions in the pronephros and genetically interacts with prickle1 in this process. Furthermore, knockdown experiments in mammalian cells demonstrate that CCDC151 is implicated in the regulation of primary cilium length. Hence, CCDC151 is required for motile cilia function in animals but has acquired additional non-motile functions in vertebrates.

Links

PubMed Online version:10.1093/hmg/ddt445

Keywords

Amino Acid Sequence; Animals; Animals, Genetically Modified; Axoneme/metabolism; Biological Transport; Cell Polarity; Cilia/genetics; Cilia/metabolism; Conserved Sequence; Drosophila/embryology; Drosophila/genetics; Drosophila Proteins/chemistry; Drosophila Proteins/genetics; Drosophila Proteins/metabolism; Embryo, Nonmammalian/cytology; Ependyma/cytology; Flagella/metabolism; Gene Expression Regulation, Developmental; Gene Knockdown Techniques; Kidney Diseases/genetics; Kidney Diseases/pathology; Mice; Phylogeny; Protein Structure, Tertiary; Proteins/chemistry; Proteins/metabolism; Zebrafish/embryology; Zebrafish/genetics; Zebrafish Proteins/chemistry; Zebrafish Proteins/genetics; Zebrafish Proteins/metabolism

Significance

Annotations

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

DANRE:A0A2R8RI11

involved_in

GO:0000132: establishment of mitotic spindle orientation

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-060209-7,ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:A0A2R8RI11

involved_in

GO:0007368: determination of left/right symmetry

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:A0A2R8RI11

involved_in

GO:0003341: cilium movement

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:A0A2R8RI11

located_in

GO:0031514: motile cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DANRE:A0A2R8RI11

located_in

GO:0036064: ciliary basal body

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DANRE:A0A2R8RI11

involved_in

GO:0070121: Kupffer's vesicle development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:A0A2R8RI11

involved_in

GO:0036159: inner dynein arm assembly

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:B3DGT8

involved_in

GO:0000132: establishment of mitotic spindle orientation

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-060209-7,ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:F1Q6J9

involved_in

GO:0000132: establishment of mitotic spindle orientation

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-060209-7,ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:F1QFD6

located_in

GO:0005929: cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DANRE:F1QFD6

located_in

GO:0036064: ciliary basal body

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DANRE:F1QFD6

involved_in

GO:0000132: establishment of mitotic spindle orientation

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-060209-7,ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:F1QFD6

involved_in

GO:0007368: determination of left/right symmetry

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:F1QFD6

involved_in

GO:0003341: cilium movement

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:F1QFD6

located_in

GO:0031514: motile cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DANRE:F1QFD6

involved_in

GO:0070121: Kupffer's vesicle development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:F1QFD6

involved_in

GO:0036159: inner dynein arm assembly

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:F1QW87

located_in

GO:0005930: axoneme

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DANRE:Q08BR0

involved_in

GO:0000132: establishment of mitotic spindle orientation

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-060209-7,ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:Q08BR0

involved_in

GO:0007368: determination of left/right symmetry

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:Q08BR0

involved_in

GO:0003341: cilium movement

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:Q08BR0

located_in

GO:0031514: motile cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DANRE:Q08BR0

located_in

GO:0036064: ciliary basal body

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DANRE:Q08BR0

involved_in

GO:0070121: Kupffer's vesicle development

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:Q08BR0

involved_in

GO:0036159: inner dynein arm assembly

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:Q5U3R2

located_in

GO:0005930: axoneme

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DANRE:Q6WGK5

involved_in

GO:0000132: establishment of mitotic spindle orientation

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-060209-7,ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

DANRE:Q7ZZC5

involved_in

GO:0000132: establishment of mitotic spindle orientation

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-060209-7,ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

MOUSE:ODAD3

located_in

GO:0005929: cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

MOUSE:ODAD3

located_in

GO:0005814: centriole

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

MOUSE:ODAD3

located_in

GO:0036064: ciliary basal body

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

MOUSE:ODAD3

involved_in

GO:1902017: regulation of cilium assembly

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

DROME:Q9VTQ3

located_in

GO:0097542: ciliary tip

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DROME:Q9VTQ3

involved_in

GO:0003341: cilium movement

ECO:0000250: sequence similarity evidence used in manual assertion

UniProtKB:F1QFD6

P

Seeded From UniProt

complete

DROME:Q9VTQ3

located_in

GO:0035253: ciliary rootlet

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DROME:Q9VTQ3

located_in

GO:0031514: motile cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DROME:Q9VTQ3

located_in

GO:0044297: cell body

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

DROME:Q9VTQ3

involved_in

GO:0048060: negative gravitaxis

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

DROME:Q9VTQ3

involved_in

GO:0070286: axonemal dynein complex assembly

ECO:0000250: sequence similarity evidence used in manual assertion

UniProtKB:F1QFD6

P

Seeded From UniProt

complete

DANRE:Q6WGK5

involved_in

GO:0000132: establishment of mitotic spindle orientation

ECO:0000316: genetic interaction evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-060209-7
ZFIN:ZDB-MRPHLNO-140225-8

P

Seeded From UniProt

complete

Notes

See also

References

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