Please use this identifier to cite or link to this item: https://doi.org/10.1096/fj.11-202663
Title: Macrophage-stimulating protein and calcium homeostasis in zebrafish
Authors: Huitema, L.F.A.
Renn, J. 
Logister, I.
Gray, J.K.
Waltz, S.E.
Flik, G.
Schulte-Merker, S.
Keywords: Bone
Mineralization
Msp
Osteogenesis
Ron
Stanniocalcin
Issue Date: Oct-2012
Source: Huitema, L.F.A., Renn, J., Logister, I., Gray, J.K., Waltz, S.E., Flik, G., Schulte-Merker, S. (2012-10). Macrophage-stimulating protein and calcium homeostasis in zebrafish. FASEB Journal 26 (10) : 4092-4101. ScholarBank@NUS Repository. https://doi.org/10.1096/fj.11-202663
Abstract: To systematically identify novel gene functions essential for osteogenesis and skeletal mineralization, we performed a forward genetic mutagenesis screen in zebrafish and isolated a mutant that showed delayed skeletal mineralization. Analysis of the mutant phenotype in an osterix:nuclear-GFP transgenic background demonstrated that mutants contain osterix-expressing osteoblasts comparable to wild-type embryos. Positional cloning revealed a premature stop mutation in the macrophage-stimulating protein (msp) gene, predicted to result in a biologically inactive protein. Analysis of the embryonic expression pattern for the receptor for Msp, Ron, shows specific expression in the corpuscles of Stannius, a teleost-specific organ that produces stanniocalcin, a pivotal hormone in fish calcium homeostasis. Knockdown of Ron resulted in identical phenotypes as observed in msp mutants. Msp mutant embryos could be rescued by excess calcium. Consistent with a role for Msp/Ron in calcium homeostasis, calcium-regulating factors, such as pth1, pth2, stc1l, and trpv5/6 were significantly affected in msp mutant larvae. While Msp and Ron have previously been shown to play a critical role in a wide variety of biological processes, we introduce here the Msp/Ron signaling axis as a previously unappreciated player in calcium homeostasis and embryonic skeletal mineralization. © FASEB.
Source Title: FASEB Journal
URI: http://scholarbank.nus.edu.sg/handle/10635/101047
ISSN: 15306860
DOI: 10.1096/fj.11-202663
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