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Novel zebrafish polycystic kidney disease models reveal functions of the hippo pathway in renal cystogenesis
Ren, Zhiqin; Zhang, Zhiwei; Liu, Tzu Ming; Ge, Wei
2021-11-01
Source PublicationDMM Disease Models and Mechanisms
ISSN1754-8403
Volume14Issue:11
Abstract

The Hippo signaling pathway is a kinase cascade that plays an important role in organ size control. As the main effectors of the Hippo pathway, transcription coactivators Yap1/Wwtr1 are regulated by the upstream kinase Stk3. Recent studies in mammals have implicated the Hippo pathway in kidney development and kidney diseases. To further illustrate its roles in vertebrate kidney, we generated a series of zebrafish mutants targeting stk3, yap1 and wwtr1 genes. The stk3−/− mutant exhibited edema, formation of glomerular cysts and pronephric tubule dilation during the larval stage. Interestingly, disruption of wwtr1, but not yap1, significantly alleviated the renal phenotypes of the stk3−/ mutant, and overexpression of Wwtr1 with the CMV promoter also induced pronephric phenotypes, similar to those of the stk3−/ mutant, during larval stage. Notably, adult fish with Wwtr1 overexpression developed phenotypes similar to those of human polycystic kidney disease (PKD). Overall, our analyses revealed roles of Stk3 and Wwtr1 in renal cyst formation. Using a pharmacological approach, we further demonstrated that Stk3-deficient zebrafish could serve as a PKD model for drug development.

KeywordHippo Signaling Pathway Kidney Development Polycystic Kidney Disease Renal Cyst Formation Zebrafish Model
DOI10.1242/dmm.049027
URLView the original
Indexed BySCIE
Language英語English
WOS Research AreaCell Biology ; Pathology
WOS SubjectCell Biology ; Pathology
WOS IDWOS:000726410400003
Scopus ID2-s2.0-85119614829
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Document TypeJournal article
CollectionFaculty of Health Sciences
Corresponding AuthorGe, Wei
AffiliationDepartment of Biomedical Sciences, Centre of Reproduction, Development and Aging (CRDA), Faculty of Health Sciences, University of Macau, Taipa, 999078, Macao
First Author AffilicationCentre of Reproduction, Development and Aging
Corresponding Author AffilicationCentre of Reproduction, Development and Aging
Recommended Citation
GB/T 7714
Ren, Zhiqin,Zhang, Zhiwei,Liu, Tzu Ming,et al. Novel zebrafish polycystic kidney disease models reveal functions of the hippo pathway in renal cystogenesis[J]. DMM Disease Models and Mechanisms,2021,14(11).
APA Ren, Zhiqin,Zhang, Zhiwei,Liu, Tzu Ming,&Ge, Wei.(2021).Novel zebrafish polycystic kidney disease models reveal functions of the hippo pathway in renal cystogenesis.DMM Disease Models and Mechanisms,14(11).
MLA Ren, Zhiqin,et al."Novel zebrafish polycystic kidney disease models reveal functions of the hippo pathway in renal cystogenesis".DMM Disease Models and Mechanisms 14.11(2021).
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