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
  • Animals
  • Body Patterning
  • Cilia/metabolism*
  • Cloning, Molecular
  • Gene Expression Profiling
  • Gene Expression Regulation, Developmental*
  • Glutathione Transferase/metabolism
  • Green Fluorescent Proteins/metabolism
  • In Situ Hybridization
  • Kidney/cytology
  • Mice
  • Models, Genetic
  • Phenotype
  • Protein Kinases/metabolism*
  • Protein Serine-Threonine Kinases/metabolism*
  • Time Factors
  • Transcription Factors/metabolism*
  • Transgenes
  • Zebrafish
  • Zebrafish Proteins/metabolism*
PubMed
22687244 Full text @ FEBS Lett.
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
Figures
Expression
Phenotype
Mutation and Transgenics
Human Disease / Model Data
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping
Errata and Notes