期刊
FEBS LETTERS
卷 586, 期 16, 页码 2273-2279出版社
ELSEVIER SCIENCE BV
DOI: 10.1016/j.febslet.2012.05.064
关键词
Primary cilia; Inv; Nek8; Zebrafish; Nephronophthisis
资金
- Ministry of Education, Culture, Sports, Science and Technology (MEXT) of Japan [21890221]
- MEXT [21790187]
- Grants-in-Aid for Scientific Research [21890221, 21790187] Funding Source: KAKEN
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.
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