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PMID:22718903

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Citation

Zhang, Q, Liu, Q, Austin, C, Drummond, I and Pierce, EA (2012) Knockdown of ttc26 disrupts ciliogenesis of the photoreceptor cells and the pronephros in zebrafish. Mol. Biol. Cell 23:3069-78

Abstract

In our effort to understand genetic disorders of the photoreceptor cells of the retina, we have focused on intraflagellar transport in photoreceptor sensory cilia. From previous mouse proteomic data we identified a cilia protein Ttc26, orthologue of dyf-13 in Caenorhabditis elegans, as a target. We localized Ttc26 to the transition zone of photoreceptor and to the transition zone of cilia in cultured murine inner medullary collecting duct 3 (mIMCD3) renal cells. Knockdown of Ttc26 in mIMCD3 cells produced shortened and defective primary cilia, as revealed by immunofluorescence and scanning electron microscopy. To study Ttc26 function in sensory cilia in vivo, we utilized a zebrafish vertebrate model system. Morpholino knockdown of ttc26 in zebrafish embryos caused ciliary defects in the pronephric kidney at 27 h postfertilization and distension/dilation of pronephros at 5 d postfertilization (dpf). In the eyes, the outer segments of photoreceptor cells appeared shortened or absent, whereas cellular lamination appeared normal in retinas at 5 dpf. This suggests that loss of ttc26 function prevents normal ciliogenesis and differentiation in the photoreceptor cells, and that ttc26 is required for normal development and differentiation in retina and pronephros. Our studies support the importance of Ttc26 function in ciliogenesis and suggest that screening for TTC26 mutations in human ciliopathies is justified.

Links

PubMed PMC3418303 Online version:10.1091/mbc.E12-01-0019

Keywords


Significance

Annotations

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

MOUSE:SSR3

located_in

GO:0005929: cilium

ECO:0000314: direct assay evidence used in manual assertion

C

  • part_of:(EMAPA:28370)

Seeded From UniProt

complete

MOUSE:CE164

located_in

GO:0005929: cilium

ECO:0000314: direct assay evidence used in manual assertion

C

  • part_of:(EMAPA:28370)

Seeded From UniProt

complete

DANRE:IFT56

acts_upstream_of_or_within

GO:0008594: photoreceptor cell morphogenesis

ECO:0000314: direct assay evidence used in manual assertion

P

Seeded From UniProt

complete

DANRE:IFT56

acts_upstream_of_or_within

GO:0060271: cilium assembly

ECO:0000314: direct assay evidence used in manual assertion

P

Seeded From UniProt

complete

DANRE:IFT56

acts_upstream_of_or_within

GO:0046530: photoreceptor cell differentiation

ECO:0000314: direct assay evidence used in manual assertion

P

Seeded From UniProt

complete

RAT:IFT56

located_in

GO:0005929: cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

MOUSE:IFT56

located_in

GO:0005929: cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

MOUSE:IFT56

located_in

GO:0005929: cilium

ECO:0000314: direct assay evidence used in manual assertion

C

  • part_of:(CL:0000210)

Seeded From UniProt

complete

MOUSE:IFT56

located_in

GO:0036064: ciliary basal body

ECO:0000314: direct assay evidence used in manual assertion

C

  • part_of:(EMAPA:28370)

Seeded From UniProt

complete

DANRE:IFT56

involved_in

GO:0060271: cilium assembly

ECO:0000314: direct assay evidence used in manual assertion

P

Seeded From UniProt

complete

DANRE:IFT56

involved_in

GO:0046530: photoreceptor cell differentiation

ECO:0000314: direct assay evidence used in manual assertion

P

Seeded From UniProt

complete

DANRE:IFT56

involved_in

GO:0008594: photoreceptor cell morphogenesis

ECO:0000314: direct assay evidence used in manual assertion

P

Seeded From UniProt

complete

DANRE:IFT56

involved_in

GO:0035845: photoreceptor cell outer segment organization

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-120906-3

P

Seeded From UniProt

complete

DANRE:IFT56

involved_in

GO:0035845: photoreceptor cell outer segment organization

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-120906-4

P

Seeded From UniProt

complete

DANRE:IFT56

involved_in

GO:0039023: pronephric duct morphogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-120906-3

P

Seeded From UniProt

complete

DANRE:IFT56

involved_in

GO:0060271: cilium assembly

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-120906-3

P

Seeded From UniProt

complete

DANRE:IFT56

involved_in

GO:0039023: pronephric duct morphogenesis

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-120906-4

P

Seeded From UniProt

complete

DANRE:IFT56

involved_in

GO:0060271: cilium assembly

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-120906-4

P

Seeded From UniProt

complete

DANRE:IFT56

involved_in

GO:0060271: cilium assembly

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete

MOUSE:IFT56

acts_upstream_of_or_within

GO:0060271: cilium assembly

ECO:0000266: sequence orthology evidence used in manual assertion

UniProtKB:Q5PR66

P

Seeded From UniProt

complete

MOUSE:IFT56

acts_upstream_of_or_within

GO:0046530: photoreceptor cell differentiation

ECO:0000266: sequence orthology evidence used in manual assertion

UniProtKB:Q5PR66

P

Seeded From UniProt

complete

MOUSE:IFT56

part_of

GO:0036064: ciliary basal body

ECO:0000314: direct assay evidence used in manual assertion

C

part_of:(EMAPA:28370)

Seeded From UniProt

complete

MOUSE:IFT56

acts_upstream_of_or_within

GO:0008594: photoreceptor cell morphogenesis

ECO:0000266: sequence orthology evidence used in manual assertion

UniProtKB:Q5PR66

P

Seeded From UniProt

complete

MOUSE:IFT56

part_of

GO:0005929: cilium

ECO:0000314: direct assay evidence used in manual assertion

C

part_of:(CL:0000210)

Seeded From UniProt

complete

MOUSE:IFT56

part_of

GO:0005929: cilium

ECO:0000314: direct assay evidence used in manual assertion

C

Seeded From UniProt

complete

MOUSE:IFT56

involved_in

GO:0060271: cilium assembly

ECO:0000315: mutant phenotype evidence used in manual assertion

P

Seeded From UniProt

complete


See also

References

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