A defect in a novel Nek-family kinase causes cystic kidney disease in the mouse and in zebrafish

S Liu, W Lu, T Obara, S Kuida, J Lehoczky, K Dewar… - 2002 - journals.biologists.com
S Liu, W Lu, T Obara, S Kuida, J Lehoczky, K Dewar, IA Drummond, DR Beier
2002journals.biologists.com
The 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 …
The murine autosomal recessive juvenile cystic kidney (jck)mutation results in polycystic kidney disease. We have identified injck mice a mutation in Nek8, a novel and highly conserved member of the Nek kinase family. In vitro expression of mutated Nek8results 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 ofNek8 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.
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