|ZFIN ID: ZDB-PUB-111207-3|
Centrin depletion causes cyst formation and other ciliopathy-related phenotypes in zebrafish
Delaval, B., Covassin, L., Lawson, N.D., and Doxsey, S.
|Source:||Cell cycle (Georgetown, Tex.) 10(22): 3964-3972 (Journal)|
|Registered Authors:||Covassin, Laurence, Lawson, Nathan|
|PubMed:||22142866 Full text @ Cell Cycle|
Delaval, B., Covassin, L., Lawson, N.D., and Doxsey, S. (2011) Centrin depletion causes cyst formation and other ciliopathy-related phenotypes in zebrafish. Cell cycle (Georgetown, Tex.). 10(22):3964-3972.
ABSTRACTMost bona fide centrosome proteins including centrins, small calcium-binding proteins, participate in spindle function during mitosis and play a role in cilia assembly in non-cycling cells. Although the basic cellular functions of centrins have been studied in lower eukaryotes and vertebrate cells in culture, phenotypes associated with centrin depletion in vertebrates in vivo has not been directly addressed. To test this, we depleted centrin2 in zebrafish and found that it leads to ciliopathy phenotypes including enlarged pronephric tubules and pronephric cysts. Consistent with the ciliopathy phenotypes, cilia defects were observed in differentiated epithelial cells of ciliated organs such as the olfactory bulb and pronephric duct. The organ phenotypes were also accompanied by cell cycle deregulation namely mitotic delay resulting from mitotic defects. Overall, this work demonstrates that centrin2 depletion causes cilia-related disorders in zebrafish. Moreover, given the presence of both cilia and mitotic defects in the affected organs, it suggests that cilia disorders may arise from a combination of these defects.