Genetic evidence that SOST inhibits WNT signaling in the limb
Abstract
SOST is a negative regulator of bone formation, and mutations in human SOST are responsible for sclerosteosis. In addition to high bone mass, sclerosteosis patients occasionally display hand defects, suggesting that SOST may function embryonically. Here we report that overexpression of SOST leads to loss of posterior structures of the zeugopod and autopod by perturbing anterior–posterior and proximal–distal signaling centers in the developing limb. Mutant mice that overexpress SOST in combination with Grem1 and Lrp6 mutations display more severe limb defects than single mutants alone, while Sost-/- significantly rescues the Lrp6-/- skeletal phenotype, signifying that SOST gain-of-function impairs limb patterning by inhibiting the WNT signaling through LRP5/6.
- Authors:
-
- Lawrence Livermore National Lab. (LLNL), Livermore, CA (United States); Univ. of California, Berkeley, CA (United States)
- Univ. of California, Davis, CA (United States)
- Univ. of California, Berkeley, CA (United States)
- Publication Date:
- Research Org.:
- Lawrence Livermore National Laboratory (LLNL), Livermore, CA (United States)
- Sponsoring Org.:
- USDOE National Nuclear Security Administration (NNSA)
- OSTI Identifier:
- 1839869
- Report Number(s):
- LLNL-JRNL-408192
Journal ID: ISSN 0012-1606; 366998
- Grant/Contract Number:
- AC52-07NA27344
- Resource Type:
- Accepted Manuscript
- Journal Name:
- Developmental Biology
- Additional Journal Information:
- Journal Volume: 342; Journal Issue: 2; Journal ID: ISSN 0012-1606
- Publisher:
- Elsevier
- Country of Publication:
- United States
- Language:
- English
- Subject:
- Biological and medical sciences; WNT signaling; SOST; Sclerostin; Shh; Limb formation
Citation Formats
Collette, Nicole M., Genetos, Damian C., Murugesh, Deepa, Harland, Richard M., and Loots, Gabriela G. Genetic evidence that SOST inhibits WNT signaling in the limb. United States: N. p., 2010.
Web. doi:10.1016/j.ydbio.2010.03.021.
Collette, Nicole M., Genetos, Damian C., Murugesh, Deepa, Harland, Richard M., & Loots, Gabriela G. Genetic evidence that SOST inhibits WNT signaling in the limb. United States. https://doi.org/10.1016/j.ydbio.2010.03.021
Collette, Nicole M., Genetos, Damian C., Murugesh, Deepa, Harland, Richard M., and Loots, Gabriela G. Tue .
"Genetic evidence that SOST inhibits WNT signaling in the limb". United States. https://doi.org/10.1016/j.ydbio.2010.03.021. https://www.osti.gov/servlets/purl/1839869.
@article{osti_1839869,
title = {Genetic evidence that SOST inhibits WNT signaling in the limb},
author = {Collette, Nicole M. and Genetos, Damian C. and Murugesh, Deepa and Harland, Richard M. and Loots, Gabriela G.},
abstractNote = {SOST is a negative regulator of bone formation, and mutations in human SOST are responsible for sclerosteosis. In addition to high bone mass, sclerosteosis patients occasionally display hand defects, suggesting that SOST may function embryonically. Here we report that overexpression of SOST leads to loss of posterior structures of the zeugopod and autopod by perturbing anterior–posterior and proximal–distal signaling centers in the developing limb. Mutant mice that overexpress SOST in combination with Grem1 and Lrp6 mutations display more severe limb defects than single mutants alone, while Sost-/- significantly rescues the Lrp6-/- skeletal phenotype, signifying that SOST gain-of-function impairs limb patterning by inhibiting the WNT signaling through LRP5/6.},
doi = {10.1016/j.ydbio.2010.03.021},
journal = {Developmental Biology},
number = 2,
volume = 342,
place = {United States},
year = {Tue Mar 30 00:00:00 EDT 2010},
month = {Tue Mar 30 00:00:00 EDT 2010}
}
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Genomic deletion of a long-range bone enhancer misregulates sclerostin in Van Buchem disease
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journal, February 2009
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Gremlin-mediated BMP antagonism induces the epithelial-mesenchymal feedback signaling controlling metanephric kidney and limb organogenesis
journal, July 2004
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Wnt and FGF signals interact to coordinate growth with cell fate specification during limb development
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