The PCP effector Fuzzy controls cilial assembly and signaling by recruiting Rab8 and Dishevelled to the primary cilium

Zilber, Yulia; Babayeva, Sima; Seo, Jung Hwa; Liu, Jiajia; Mootin, Steven; Torban, Elena

doi:10.1091/mbc.e12-06-0437

Cited by 54 publications

(72 citation statements)

References 57 publications

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“…Mutations in these genes show both PCP and ciliogenesis phenotypes (Cui et al, 2013; Gray et al, 2009; Heydeck et al, 2009; Kim et al, 2010; Park et al, 2006; Zilber et al, 2013). In the mouse both intu and fuz mutations result in neural tube defects (Heydeck and Liu, 2011; Heydeck et al, 2009) and double mutants appear similar to single mutants (Heydeck and Liu, 2011) mirroring the lack of phenotypic additivity seen in flies (Collier et al, 2005).…”

Section: Discussionmentioning

confidence: 99%

The proteins encoded by the Drosophila Planar Polarity Effector genes inturned, fuzzy and fritz interact physically and can re-pattern the accumulation of “upstream” Planar Cell Polarity proteins

Wang

Yan

Lee

et al. 2014

Developmental Biology

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The frizzled/starry night pathway regulates planar cell polarity in a wide variety of tissues in many types of animals. It was discovered and has been most intensively studied in the Drosophila wing where it controls the formation of the array of distally pointing hairs that cover the wing. The pathway does this by restricting the activation of the cytoskeleton to the distal edge of wing cells. This results in hairs initiating at the distal edge and growing in the distal direction. All of the proteins encoded by genes in the pathway accumulate asymmetrically in wing cells. The pathway is a hierarchy with the Planar Cell Polarity (PCP) genes (aka the core genes) functioning as a group upstream of the Planar Polarity Effector (PPE) genes which in turn function as a group upstream of multiple wing hairs. Upstream proteins, such as Frizzled accumulate on either the distal and/or proximal edges of wing cells. Downstream PPE proteins accumulate on the proximal edge under the instruction of the upstream proteins. A variety of types of data support this hierarchy, however, we have found that when over expressed the PPE proteins can alter both the subcellular location and level of accumulation of the upstream proteins. Thus, the epistatic relationship is context dependent. We further show that the PPE proteins interact physically and can modulate the accumulation of each other in wing cells. We also find that over expression of Frtz results in a marked delay in hair initiation suggesting that it has a separate role/activity in regulating the cytoskeleton that is not shared by other members of the group.

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Section: Discussionmentioning

confidence: 99%

The proteins encoded by the Drosophila Planar Polarity Effector genes inturned, fuzzy and fritz interact physically and can re-pattern the accumulation of “upstream” Planar Cell Polarity proteins

Wang

Yan

Lee

et al. 2014

Developmental Biology

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“…For example, a hypermorphic allele of the canonical Wnt coreceptor lipoprotein receptor-related protein, Lrp6, increased Wnt canonical and, simultaneously, abolished PCP-induced JNK activities [117]. Several proteins have been shown to act as molecular switches between the Wnt/ -catenin and PCP signaling pathways [116,118] lending credibility to this mechanism.…”

Section: Planar Cell Polarity Pathway and Kidneymentioning

confidence: 99%

“…Indeed, several recent publications presented convincing data documenting antagonistic relationship between Wnt/ -catenin activity and PCP signaling [117,118]. The inv/inv mouse has cystic kidneys and laterality defects (situs inversus) [141], and knockdown of inv in zebrafish leads to a shortened anterior-posterior axis and widened somites, a classical manifestation of defective PCP signaling.…”

Section: Advances In Nephrologymentioning

confidence: 99%

Planar Cell Polarity Pathway in Kidney Development and Function

Rocque

Torban

2015

Advances in Nephrology

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The evolutionarily conserved planar cell polarity (PCP) signaling pathway controls tissue polarity within the plane orthogonal to the apical-basal axis. PCP was originally discovered in Drosophila melanogaster where it is required for the establishment of a uniform pattern of cell structures and appendages. In vertebrates, including mammals, the PCP pathway has been adapted to control various morphogenetic processes that are critical for tissue and organ development. These include convergent extension (crucial for neural tube closure and cochlear duct development) and oriented cell division (needed for tubular elongation), ciliary tilting that enables directional fluid flow, and other processes. Recently, strong evidence has emerged to implicate the PCP pathway in vertebrate kidney development. In this review, we will describe the experimental data revealing the role of PCP signaling in nephrogenesis and kidney disease.

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“…32,35,197 However, many Wnt signaling components, such as Fz3, Dvl, and β-catenin, localize to the ciliary/centrosomal axis, and dysregulated Wnt signaling has been reported in cells and tissues with disrupted ciliogenesis or basal body integrity. [198][199][200] Moreover, in the mouse inner ear, establishment of PCP was shown to depend on the formation of functional primary cilia. 201 However, other studies in mice and zebrafish reveal no obvious canonical or non-canonical Wnt phenotypes in cilia mutants, 202,203 emphasizing the importance of further studies in this area.…”

Section: Cardiac Primary Cilia and Other Signaling Pathwaysmentioning

confidence: 99%