PUBLICATION
The ciliary protein Nek8/Nphp9 acts downstream of Inv/Nphp2 during pronephros morphogenesis and left-right establishment in zebrafish
- Authors
- Fukui, H., Shiba, D., Asakawa, K., Kawakami, K., and Yokoyama, T.
- ID
- ZDB-PUB-120628-4
- Date
- 2012
- Source
- FEBS letters 586(16): 2273-2279 (Journal)
- Registered Authors
- Kawakami, Koichi
- Keywords
- primary cells, inv, nek8, zebrafish, nephronophthisis
- MeSH Terms
-
- Mice
- Cilia/metabolism*
- Zebrafish Proteins/metabolism*
- Green Fluorescent Proteins/metabolism
- Time Factors
- Gene Expression Regulation, Developmental*
- Models, Genetic
- Protein Kinases/metabolism*
- Gene Expression Profiling
- Animals
- Kidney/cytology
- Phenotype
- In Situ Hybridization
- Zebrafish
- Glutathione Transferase/metabolism
- Cloning, Molecular
- Protein Serine-Threonine Kinases/metabolism*
- Transgenes
- Body Patterning
- Transcription Factors/metabolism*
- PubMed
- 22687244 Full text @ FEBS Lett.
Citation
Fukui, H., Shiba, D., Asakawa, K., Kawakami, K., and Yokoyama, T. (2012) The ciliary protein Nek8/Nphp9 acts downstream of Inv/Nphp2 during pronephros morphogenesis and left-right establishment in zebrafish. FEBS letters. 586(16):2273-2279.
Abstract
Nephronophthisis (NPHP) is an autosomal recessive cystic kidney disease. Among 12 reported Nphp gene products, Inv/Nphp2, Nphp3 and Nek8/Nphp9 are localized to the proximal segment in the primary cilium. However, the functional relationships are unknown. This study focused on phenotype analysis of nek8 knockdown embryos and the genetic relationship between nek8 and inv in zebrafish. Knockdown of nek8 produced both pronephric cysts and abnormal cardiac looping. Simultaneous knockdown of nek8 and inv synergistically increased the incidence of these defects. Interestingly, nek8 mRNA rescued inv morphant phenotypes, although inv mRNA could not rescue nek8 morphant phenotypes. These results suggest that Nek8 acts downstream of Inv function.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping