Centrin depletion causes cyst formation and other ciliopathy-related phenotypes in zebrafish

UMMS Affiliation

Program in Gene Function and Expression; Program in Molecular Medicine

Publication Date


Document Type



Animals; Calcium-Binding Proteins; Cilia; Embryo, Nonmammalian; Embryonic Development; Mitosis; Morpholinos; Phenotype; Zebrafish; Zebrafish Proteins


Genetics and Genomics


Most 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.

DOI of Published Version



Cell Cycle. 2011 Nov 15;10(22):3964-72. Epub 2011 Nov 15. Link to article on publisher's site

Journal/Book/Conference Title

Cell cycle (Georgetown, Tex.)

Related Resources

Link to Article in PubMed

PubMed ID