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Deficiency of Sprouty Proteins in Mice Resemble Ciliopathic Phenotype

The result's identifiers

  • Result code in IS VaVaI

    <a href="https://www.isvavai.cz/riv?ss=detail&h=RIV%2F00216305%3A26620%2F19%3APU132816" target="_blank" >RIV/00216305:26620/19:PU132816 - isvavai.cz</a>

  • Alternative codes found

    RIV/00216224:14110/19:00112474

  • Result on the web

    <a href="https://onlinelibrary.wiley.com/doi/abs/10.1002/jmor.21003" target="_blank" >https://onlinelibrary.wiley.com/doi/abs/10.1002/jmor.21003</a>

  • DOI - Digital Object Identifier

Alternative languages

  • Result language

    angličtina

  • Original language name

    Deficiency of Sprouty Proteins in Mice Resemble Ciliopathic Phenotype

  • Original language description

    The Sprouty family of proteins is a highly conserved group of intracellular modulators of receptor tyrosine kinase signaling, including the fibroblast growth factor receptor (FGFR) pathway. There are four orthologs (Sprouty1-4) in mammals. During embryonic development and postnatal bone remodeling, tight regulation of FGFR signaling is essential in endochondral ossification and in bone and mineral homeostasis. Sprouty2 was shown to be implicated in endochondral bone formation in Sprouty2-/- mice with reduced postnatal skeletal size and trabecular bone mass. Nevertheless, the underlying mechanism of its action at the molecular level remains to be elucidated. Moreover, long bones of Sprouty4-/- display limb malformations, such as chondroma-like lesions in locations adjacent to growth plate, or fused and/or supernumerary digits on front limbs. Our skeletal and other phenotypic findings on Sprouty2- and 4-deficient mice and Sprouty2/4 double knockouts (DKOs) strikingly resemble the phenotype of model mice

  • Czech name

  • Czech description

Classification

  • Type

    O - Miscellaneous

  • CEP classification

  • OECD FORD branch

    10605 - Developmental biology

Result continuities

  • Project

    Result was created during the realization of more than one project. More information in the Projects tab.

  • Continuities

    P - Projekt vyzkumu a vyvoje financovany z verejnych zdroju (s odkazem do CEP)

Others

  • Publication year

    2019

  • Confidentiality

    S - Úplné a pravdivé údaje o projektu nepodléhají ochraně podle zvláštních právních předpisů