| RRC ID |
82802
|
| Author |
Fukui H, Shiba D, Asakawa K, Kawakami K, Yokoyama T.
|
| Title |
The ciliary protein Nek8/Nphp9 acts downstream of Inv/Nphp2 during pronephros morphogenesis and left-right establishment in zebrafish.
|
| Journal |
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.
|
| Volume |
586(16)
|
| Pages |
2273-9
|
| Published |
2012-7-30
|
| DOI |
10.1016/j.febslet.2012.05.064
|
| PII |
S0014-5793(12)00463-2
|
| PMID |
22687244
|
| MeSH |
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
NIMA-Related Kinases
Phenotype
Protein Kinases / metabolism*
Protein Serine-Threonine Kinases / metabolism*
Time Factors
Transcription Factors / metabolism*
Transgenes
Zebrafish
Zebrafish Proteins / metabolism*
|
| IF |
3.057
|
| Resource |
| Zebrafish |
hspGGFF20A
UAS:GFP |
