|ZFIN ID: ZDB-PUB-021112-4|
A defect in a novel Nek-family kinase causes cystic kidney disease in the mouse and in zebrafish
Liu, S., Lu, W., Obara, T., Kuida, S., Lehoczky, J., Dewar, K., Drummond, I.A., and Beier, D.R.
|Source:||Development (Cambridge, England) 129(24): 5839-5846 (Journal)|
|Registered Authors:||Beier, David R., Drummond, Iain, Obara, Tomoko|
|Keywords:||PKD, mouse models, zebrafish, Nek kinase|
|PubMed:||12421721 Full text @ Development|
Liu, S., Lu, W., Obara, T., Kuida, S., Lehoczky, J., Dewar, K., Drummond, I.A., and Beier, D.R. (2002) A defect in a novel Nek-family kinase causes cystic kidney disease in the mouse and in zebrafish. Development (Cambridge, England). 129(24):5839-5846.
ABSTRACTThe murine autosomal recessive juvenile cystic kidney (jck) mutation results in polycystic kidney disease. We have identified in jck mice a mutation in Nek8, a novel and highly conserved member of the Nek kinase family. In vitro expression of mutated Nek8 results in enlarged, multinucleated cells with an abnormal actin cytoskeleton. To confirm that a defect in the Nek8 gene can cause cystic disease, we performed a cross-species analysis: injection of zebrafish embryos with a morpholino anti-sense oligonucleotide corresponding to the ortholog of Nek8 resulted in the formation of pronephric cysts. These results demonstrate that comparative analysis of gene function in different model systems represents a powerful means to annotate gene function.