TALEN-mediated mutagenesis in zebrafish reveals a role for r-spondin 2 in fin ray and vertebral development

•Null-type and hypomorphic rspo2 zebrafish mutant lines were generated.•Hypomorphic mutant lacked C-terminal thrombospondin domain.•Null mutants did not form fin ray skeletons, but hypomorphic mutants developed them normally.•Both mutants exhibited hypoplasia of the neural/hemal arches and ribs.•Rsp...

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Bibliographic Details
Published in:FEBS letters 2014-12, Vol.588 (24), p.4543-4550
Main Authors: Tatsumi, Yoshiaki, Takeda, Moe, Matsuda, Masaru, Suzuki, Tohru, Yokoi, Hayato
Format: Article
Language:English
Subjects:
Amino Acid Sequence
Animals
Danio rerio
DNA Restriction Enzymes - metabolism
Fin ray
fins
Gene Expression Regulation
Humans
Intercellular Signaling Peptides and Proteins - chemistry
Intercellular Signaling Peptides and Proteins - genetics
Intercellular Signaling Peptides and Proteins - metabolism
Molecular Sequence Data
musculoskeletal system
Mutagenesis
mutants
Mutation
Protein Structure, Tertiary
R-spondin2
Skates (Fish) - abnormalities
Skates (Fish) - genetics
Skates (Fish) - growth & development
Skeletogenesis
TALEN
Zebrafish
Zebrafish - genetics
Zebrafish Proteins - chemistry
Zebrafish Proteins - genetics
Zebrafish Proteins - metabolism
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Summary:•Null-type and hypomorphic rspo2 zebrafish mutant lines were generated.•Hypomorphic mutant lacked C-terminal thrombospondin domain.•Null mutants did not form fin ray skeletons, but hypomorphic mutants developed them normally.•Both mutants exhibited hypoplasia of the neural/hemal arches and ribs.•Rspo2 mutants are useful for characterization of Rspo2 function. 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, rspo2null, lacking all functional domains, and a hypomorphic mutant, rspo2tsp, lacking the two N-terminal domains. Mutants were analyzed mainly for abnormalities in the skeletal system. Fin ray skeletons were formed normally in the rspo2tsp mutants, but were absent from the rspo2null 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.
ISSN:0014-5793
1873-3468
DOI:10.1016/j.febslet.2014.10.015