Null and hypomorph Prickle1 alleles in mice phenocopy human Robinow syndrome and disrupt signaling downstream of Wnt5a

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Null and hypomorph Prickle1 alleles in mice phenocopy human Robinow syndrome and disrupt signaling downstream of Wnt5a is …
instance of (P31):
scholarly articleQ13442814

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P356DOI10.1242/BIO.20148375
P932PMC publication ID4163663
P698PubMed publication ID25190059
P5875ResearchGate publication ID267026916

P50authorTiansen LiQ83229635
Anand SwaroopQ30506132
P2093author name stringChen Lin
Chunqiao Liu
Chun Gao
Helen May-Simera
P2860cites workRegulation of planar cell polarity by Smurf ubiquitin ligasesQ24317525
Robinow syndromeQ24679335
Mutation of Celsr1 disrupts planar polarity of inner ear hair cells and causes severe neural tube defects in the mouseQ28184694
Convergent extension: the molecular control of polarized cell movement during embryonic developmentQ28204202
Planar polarity from flies to vertebratesQ28239429
Diego and Prickle regulate Frizzled planar cell polarity signalling by competing for Dishevelled bindingQ28254777
Nuclear localization of Prickle2 is required to establish cell polarity during early mouse embryogenesisQ28259767
Prickle1 is expressed in distinct cell populations of the central nervous system and contributes to neuronal morphogenesisQ28285792
Mutations in VANGL1 associated with neural-tube defectsQ28296545
Inversin, a novel gene in the vertebrate left-right axis pathway, is partially deleted in the inv mouseQ28504917
A Wnt5a pathway underlies outgrowth of multiple structures in the vertebrate embryoQ28505232
Wnt5a functions in planar cell polarity regulation in miceQ28505898
Dishevelled 2 is essential for cardiac outflow tract development, somite segmentation and neural tube closureQ28507430
Wnt9b signaling regulates planar cell polarity and kidney tubule morphogenesisQ28510389
Murine dishevelled 3 functions in redundant pathways with dishevelled 1 and 2 in normal cardiac outflow tract, cochlea, and neural tube developmentQ28511382
Lack of cadherins Celsr2 and Celsr3 impairs ependymal ciliogenesis, leading to fatal hydrocephalusQ28511503
Frizzled 1 and frizzled 2 genes function in palate, ventricular septum and neural tube closure: general implications for tissue fusion processesQ28586361
The planar cell polarity gene Vangl2 is required for mammalian kidney-branching morphogenesis and glomerular maturationQ28586682
Wnt proteins induce dishevelled phosphorylation via an LRP5/6- independent mechanism, irrespective of their ability to stabilize beta-cateninQ28587260
Asymmetric distribution of prickle-like 2 reveals an early underlying polarization of vestibular sensory epithelia in the inner earQ28587932
Induced Wnt5a expression perturbs embryonic outgrowth and intestinal elongation, but is well-tolerated in adult mice.Q50802085
Novel Robinow syndrome causing mutations in the proximal region of the frizzled-like domain of ROR2 are retained in the endoplasmic reticulum.Q51979665
The left-right determinant Inversin is a component of node monocilia and other 9+0 cilia.Q52107835
Linkage of recessive Robinow syndrome to a 4 cM interval on chromosome 9q22Q57320079
Examining planar cell polarity in the mammalian cochleaQ83179154
Severe neural tube defects in the loop-tail mouse result from mutation of Lpp1, a novel gene involved in floor plate specificationQ28591062
Identification of Vangl2 and Scrb1 as planar polarity genes in mammalsQ28593071
The role of Frizzled3 and Frizzled6 in neural tube closure and in the planar polarity of inner-ear sensory hair cellsQ28593180
Inversin, the gene product mutated in nephronophthisis type II, functions as a molecular switch between Wnt signaling pathwaysQ29614619
Robinow syndrome: report of two patients and review of literatureQ33860252
Wnt5a can both activate and repress Wnt/β-catenin signaling during mouse embryonic developmentQ34286592
Wnt signaling gradients establish planar cell polarity by inducing Vangl2 phosphorylation through Ror2Q34706774
Wnt5a-Ror-Dishevelled signaling constitutes a core developmental pathway that controls tissue morphogenesisQ35837144
Regulation of retinal progenitor expansion by Frizzled receptors: implications for microphthalmia and retinal colobomaQ35856253
Planar cell polarity, ciliogenesis and neural tube defectsQ36599301
Planar cell polarity signaling: a common mechanism for cellular polarization.Q36606892
Frizzled/PCP signalling: a conserved mechanism regulating cell polarity and directed motilityQ36710748
Tissue/planar cell polarity in vertebrates: new insights and new questionsQ36720615
Wg and Wnt4 provide long-range directional input to planar cell polarity orientation in Drosophila.Q37146876
Mouse prickle1, the homolog of a PCP gene, is essential for epiblast apical-basal polarityQ37321035
Wnt signaling pathways meet Rho GTPases.Q37388913
Asymmetric protein localization in planar cell polarity: mechanisms, puzzles, and challenges.Q38058902
Prickle 1 regulates cell movements during gastrulation and neuronal migration in zebrafishQ38352359
Zebrafish prickle, a modulator of noncanonical Wnt/Fz signaling, regulates gastrulation movements.Q38520593
Prickle1 stunts limb growth through alteration of cell polarity and gene expressionQ38728039
Neural tube closure requires Dishevelled-dependent convergent extension of the midlineQ39750691
The prickle-related gene in vertebrates is essential for gastrulation cell movementsQ39750801
Microtubules enable the planar cell polarity of airway cilia.Q41825907
Disheveled mediated planar cell polarity signaling is required in the second heart field lineage for outflow tract morphogenesisQ42322638
When whorls collide: the development of hair patterns in frizzled 6 mutant miceQ42375659
Disruption of PCP signaling causes limb morphogenesis and skeletal defects and may underlie Robinow syndrome and brachydactyly type B.Q42586197
Ltap, a mammalian homolog of Drosophila Strabismus/Van Gogh, is altered in the mouse neural tube mutant Loop-tailQ42653433
Alcian blue/alizarin red staining of cartilage and bone in mouse.Q43166098
An essential role for Frizzled5 in neuronal survival in the parafascicular nucleus of the thalamus.Q45914648
frizzled regulates mirror-symmetric pattern formation in the Drosophila eye.Q47071507
Recessive Robinow syndrome, allelic to dominant brachydactyly type B, is caused by mutation of ROR2.Q47839446
P275copyright licenseCreative Commons Attribution 3.0 UnportedQ14947546
P6216copyright statuscopyrightedQ50423863
P4510describes a project that usesImageJQ1659584
P433issue9
P407language of work or nameEnglishQ1860
P921main subjectRobinow syndromeQ1475743
P304page(s)861-870
P577publication date2014-09-04
P1433published inBiology OpenQ27724268
P1476titleNull and hypomorph Prickle1 alleles in mice phenocopy human Robinow syndrome and disrupt signaling downstream of Wnt5a
P478volume3

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cites work (P2860)
Q38697548A proteomic analysis of LRRK2 binding partners reveals interactions with multiple signaling components of the WNT/PCP pathway
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Q36245053Control of vertebrate core planar cell polarity protein localization and dynamics by Prickle 2.
Q42766415Mesp1 controls the speed, polarity, and directionality of cardiovascular progenitor migration.
Q39450241Molecular genetics and targeted therapy of WNT-related human diseases (Review).
Q35589023Mutations in DVL1 cause an osteosclerotic form of Robinow syndrome.
Q59104245Planar Cell Polarity Pathway in Kidney Development and Function
Q36736933Prickle1 mutation causes planar cell polarity and directional cell migration defects associated with cardiac outflow tract anomalies and other structural birth defects.
Q60300469Prickle1 regulates differentiation of frontal bone osteoblasts
Q41512358Prickle1 regulates neurite outgrowth of apical spiral ganglion neurons but not hair cell polarity in the murine cochlea
Q36785209Prickle3 synergizes with Wtip to regulate basal body organization and cilia growth.
Q63681658Robinow syndrome skeletal phenotypes caused by the WNT5AC83S variant are due to dominant interference with chondrogenesis
Q96220331Shear stimulation of FOXC1 and FOXC2 differentially regulates cytoskeletal activity during lymphatic valve maturation
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