FEBS Letters · 2014 · 26 citations · 29 references
Life SciencesDevelopmental BiologyGeneticsEmbryogenesisMorphogenesisOrganogenesisCraniofacial Developmental GeneticsDorsal-ventral Axis FormationFish Developmental BiologySkeletal DevelopmentCanonical Wnt PathwayFunctional Analysis Of Protein DomainsKnockout Zebrafish ModelsMorphogen Signaling In AppendagesMutations In Wnt Signaling PathwaySkeletal System AnatomyVertebral ArchesWnt Signaling Modulation
R-spondin (Rspo) encodes a multi-domain protein that modulates the Wnt-signaling pathway. Two distinct rspo2 zebrafish mutants were generated by TALEN-mediated mutagenesis: a null mutant, rspo2(null), lacking all functional domains, and a hypomorphic mutant, rspo2(tsp), lacking the two N-terminal domains. Mutants were analyzed mainly for abnormalities in the skeletal system. Fin ray skeletons were formed normally in the rspo2(tsp) mutants, but were absent from the rspo2(null) mutants. Hypoplasia of the neural/hemal arches and ribs was observed in both mutants. Thus, the two rspo2 mutants help to identify the functions of Rspo2 in skeletogenesis, as well as functional differences among multiple Rspo2 domains.
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LGR4 and LGR5 are R‐spondin receptors mediating Wnt/β‐catenin and Wnt/PCP signalling
Andrei Glinka, Christine Dolde, Nadine Kirsch et al. · EMBO Reports · 2011 · 566 citations · Full text