The planar cell polarity effector Fuz is essential for targeted membrane trafficking, ciliogenesis and mouse embryonic development

Gray, Ryan S.; Abitua, Philip B.; Wlodarczyk, Bogdan J.; Szabo-Rogers, Heather L.; Blanchard, Otis; Lee, Insuk; Weiss, Greg S.; Liu, Karen; Marcotte, Edward M.; Wallingford, John B.; Finnell, Richard H.

doi:10.1038/ncb1966

Cited by 205 publications

(267 citation statements)

References 46 publications

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“…Loss of their activity in frog and in mouse has only mild effects on gastrulation, but disrupts ciliogenesis, additionally causing defects in Hh signalling due to cilia loss (Park et al, 2006;Gray et al, 2009;Heydeck et al, 2009;Kim et al, 2010;Zeng et al, 2010). Furthermore, during frog gastrulation, Frtz loss affects the ability of cells to elongate but not their polarity (Kim et al, 2010).…”

Section: Downstream Effectorsmentioning

confidence: 99%

Principles of planar polarity in animal development

2011

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SummaryPlanar polarity describes the coordinated polarisation of cells or structures in the plane of a tissue. The patterning mechanisms that underlie planar polarity are well characterised in Drosophila, where many events are regulated by two pathways: the 'core' planar polarity complex and the Fat/Dachsous system. Components of both pathways also function in vertebrates and are implicated in diverse morphogenetic processes, some of which self-evidently involve planar polarisation and some of which do not. Here, we review the molecular mechanisms and cellular consequences of planar polarisation in diverse contexts, seeking to identify the common principles across the animal kingdom.

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Section: Downstream Effectorsmentioning

confidence: 99%

Principles of planar polarity in animal development

2011

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“…In contrast to the Wnt/-catenin pathway that transcriptionally regulates foxj1a, PCP effectors and core proteins regulate cell and basal body polarization (Oishi et al, 2006;Park, T. J. et al, 2006;Park et al, 2008;Gray et al, 2009;Mitchell et al, 2009;Borovina et al, 2010;Guirao et al, 2010;Song et al, 2010). In fact, ligands and receptors that participate in these two pathways are colocalized in many ciliated tissues.…”

Section: ; Elmentioning

confidence: 99%

Wnt/β-catenin signaling directly regulates Foxj1 expression and ciliogenesis in zebrafish Kupffer’s vesicle

2012

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SUMMARYCilia are essential for normal development. The composition and assembly of cilia has been well characterized, but the signaling and transcriptional pathways that govern ciliogenesis remain poorly studied. Here, we report that Wnt/-catenin signaling directly regulates ciliogenic transcription factor foxj1a expression and ciliogenesis in zebrafish Kupffer's vesicle (KV). We show that Wnt signaling acts temporally and KV cell-autonomously to control left-right (LR) axis determination and ciliogenesis. Specifically, reduction of Wnt signaling leads to a disruption of LR patterning, shorter and fewer cilia, a loss of cilia motility and a downregulation of foxj1a expression. However, these phenotypes can be rescued by KV-targeted overexpression of foxj1a. In comparison to the FGF pathway that has been previously implicated in the control of ciliogenesis, our epistatic studies suggest a more downstream function of Wnt signaling in the regulation of foxj1a expression and ciliogenesis in KV. Importantly, enhancer analysis reveals that KV-specific expression of foxj1a requires the presence of putative Lef1/Tcf binding sites, indicating that Wnt signaling activates foxj1a transcription directly. We also find that impaired Wnt signaling leads to kidney cysts and otolith disorganization, which can be attributed to a loss of foxj1 expression and disrupted ciliogenesis in the developing pronephric ducts and otic vesicles. Together, our data reveal a novel role of Wnt/-catenin signaling upstream of ciliogenesis, which might be a general developmental mechanism beyond KV. Moreover, our results also prompt a hypothesis that certain developmental effects of the Wnt/-catenin pathway are due to the activation of Foxj1 and cilia formation. KEY WORDS: Wnt/-catenin signaling, Ciliogenesis, Foxj1, Kupffer's vesicle ZebrafishWnt/-catenin signaling directly regulates Foxj1 expression and ciliogenesis in zebrafish Kupffer's vesicle

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“…For example, Rab8 is a regulator of vesicular trafficking and it mediates the Bardet-Biedl symdrome (BBS) protein complex regulation of ciliogenesis [5,11]. Fuz, a planar cell polarity effector protein, also regulates vesicular trafficking and ciliogenesis [12]. Furthermore, a recent genome-wide RNAi screen identifies proteins of the endocytic recycling pathway as modulators of ciliogenesis [13].…”

Section: Introductionmentioning

confidence: 99%

A SNX10/V-ATPase pathway regulates ciliogenesis in vitro and in vivo

Chen

et al. 2011

Cell Res

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Sorting nexins (SNXs) are phosphoinositide-binding proteins implicated in the sorting of various membrane proteins in vitro, but the in vivo functions of them remain largely unknown. We reported previously that SNX10 is a unique member of the SNX family genes in that it has vacuolation activity in cells. We investigate the biological function of SNX10 by loss-of-function assay in this study and demonstrate that SNX10 is required for the formation of primary cilia in cultured cells. In zebrafish, SNX10 is involved in ciliogenesis in the Kupffer's vesicle and essential for left-right patterning of visceral organs. Mechanistically, SNX10 interacts with V-ATPase complex and targets it to the centrosome where ciliogenesis is initiated. Like SNX10, V-ATPase regulates ciliogenesis in vitro and in vivo and does so synergistically with SNX10. We further discover that SNX10 and V-ATPase regulate the ciliary trafficking of Rab8a, which is a critical regulator of ciliary membrane extension. These results identify an SNX10/V-ATPaseregulated vesicular trafficking pathway that is crucial for ciliogenesis, and reveal that SNX10/V-ATPase, through the regulation of cilia formation in various organs, play an essential role during early embryonic development.

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The planar cell polarity effector Fuz is essential for targeted membrane trafficking, ciliogenesis and mouse embryonic development

Cited by 205 publications

References 46 publications

Principles of planar polarity in animal development

Principles of planar polarity in animal development

Wnt/β-catenin signaling directly regulates Foxj1 expression and ciliogenesis in zebrafish Kupffer’s vesicle

A SNX10/V-ATPase pathway regulates ciliogenesis in vitro and in vivo

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