PUBLICATION
A defect in a novel Nek-family kinase causes cystic kidney disease in the mouse and in zebrafish
- Authors
- Liu, S., Lu, W., Obara, T., Kuida, S., Lehoczky, J., Dewar, K., Drummond, I.A., and Beier, D.R.
- ID
- ZDB-PUB-021112-4
- Date
- 2002
- 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
- MeSH Terms
-
- Amino Acid Sequence
- Animals
- COS Cells
- Cell Nucleus/metabolism
- Cloning, Molecular
- Databases as Topic
- Disease Models, Animal
- Genome
- Kidney Diseases, Cystic/genetics*
- Mice
- Mice, Inbred C57BL
- Models, Genetic
- Molecular Sequence Data
- Mutation
- Oligonucleotides, Antisense/pharmacology
- Physical Chromosome Mapping
- Protein Kinases*
- Protein Serine-Threonine Kinases/genetics*
- Protein Serine-Threonine Kinases/physiology*
- RNA, Messenger/metabolism
- Reverse Transcriptase Polymerase Chain Reaction
- Time Factors
- Zebrafish
- Zebrafish Proteins
- PubMed
- 12421721 Full text @ Development
Citation
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.
Abstract
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 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.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping