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Publication : A conditional allele of Rspo3 reveals redundant function of R-spondins during mouse limb development.

First Author  Neufeld S Year  2012
Journal  Genesis Volume  50
Issue  10 Pages  741-9
PubMed ID  22610508 Mgi Jnum  J:188812
Mgi Id  MGI:5442257 Doi  10.1002/dvg.22040
Citation  Neufeld S, et al. (2012) A conditional allele of Rspo3 reveals redundant function of R-spondins during mouse limb development. Genesis 50(10):741-9
abstractText  Summary: R-spondins are secreted ligands that bind cell surface receptors and activate Wnt/beta-catenin signaling. Human mutations and gene inactivation studies in mice have revealed a role for these four proteins (RSPO1-4) in diverse developmental processes ranging from sex determination to limb development. Among the genes coding for R-spondins, only inactivation of Rspo3 shows early embryonic lethality (E10.5 in mice). Therefore, a conditional allele of this gene is necessary to understand the function of R-spondins throughout murine development. To address this need, we have produced an allele in which loxP sites flank exons 2-4 of Rspo3, allowing tissue-specific deletion of these exons in the presence of Cre recombinase. We used these mice to investigate the role of Rspo3 during limb development and found that limbs ultimately developed normally in the absence of Rspo3 function. However, severe hindlimb truncations resulted when Rspo3 and Rspo2 mutations were combined, demonstrating redundant function of these genes. genesis 50:741-749, 2012. (c) 2012 Wiley Periodicals, Inc.
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