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PMID:26327644

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Citation

Dutta, S, Sriskanda, S, Boobalan, E, Alur, RP, Elkahloun, A and Brooks, BP (2015) nlz1 is required for cilia formation in zebrafish embryogenesis. Dev. Biol. 406:203-11

Abstract

The formation of cilia is a fundamental developmental process affecting diverse functions such as cellular signaling, tissue morphogenesis and body patterning. However, the mechanisms of ciliogenesis during vertebrate development are not fully understood. In this report we describe a novel role of the Nlz1 protein in ciliogenesis. We demonstrate morpholino-mediated knockdown of nlz1 in zebrafish causes abnormal specification of the cells of Kupffer's vesicle (KV); a severe reduction of the number of cilia in KV, the pronephros, and the neural floorplate; and a spectrum of later phenotypes reminiscent of human ciliopathies. In vitro and in vivo data indicate that Nlz1 acts downstream of Foxj1a and Wnt8a/presumed canonical Wnt signaling. Furthermore, Nlz1 contributes to motile cilia formation by positively regulating Wnt11/presumed non-canonical Wnt signaling. Together, our data suggest a novel role of nlz1 in ciliogenesis and the morphogenesis of multiple tissues.

Links

PubMed PMC4639414 Online version:10.1016/j.ydbio.2015.08.019

Keywords

Alcian Blue; Animals; Blotting, Western; Cilia/physiology; DNA Primers/genetics; DNA-Binding Proteins/genetics; DNA-Binding Proteins/metabolism; Forkhead Transcription Factors/metabolism; Gene Expression Regulation, Developmental/physiology; In Situ Hybridization; Luciferases; Microscopy, Confocal; Morphogenesis/physiology; Real-Time Polymerase Chain Reaction; Reverse Transcriptase Polymerase Chain Reaction; Wnt Proteins/metabolism; Wnt Signaling Pathway/physiology; Zebrafish/embryology; Zebrafish Proteins/genetics; Zebrafish Proteins/metabolism

Significance

Annotations

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

DANRE:FXJ1A

involved_in

GO:0007368: determination of left/right symmetry

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-090409-2

P

Seeded From UniProt

complete

DANRE:FXJ1A

involved_in

GO:0060271: cilium assembly

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-090409-2

P

Seeded From UniProt

complete

DANRE:ZN703

involved_in

GO:0007368: determination of left/right symmetry

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-090413-3

P

Seeded From UniProt

complete

DANRE:ZN703

involved_in

GO:0060271: cilium assembly

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-090413-3

P

Seeded From UniProt

complete

DANRE:Q08CI2

involved_in

GO:0007368: determination of left/right symmetry

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-090409-2

P

Seeded From UniProt

complete

DANRE:Q08CI2

involved_in

GO:0060271: cilium assembly

ECO:0000315: mutant phenotype evidence used in manual assertion

ZFIN:ZDB-MRPHLNO-090409-2

P

Seeded From UniProt

complete

Notes

See also

References

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