Sost and its paralog Sostdc1 coordinate digit number in a Gli3-dependent manner
Abstract
WNT signaling is critical in most aspects of skeletal development and homeostasis, and antagonists of WNT signaling are emerging as key regulatory proteins with great promise as therapeutic agents for bone disorders. Here we show that Sost and its paralog Sostdc1 emerged through ancestral genome duplication and their expression patterns have diverged to delineate nonoverlapping domains in most organ systems including musculoskeletal, cardiovascular, nervous, digestive, reproductive and respiratory. In the developing limb, Sost and Sostdc1 display dynamic expression patterns with Sost being restricted to the distal ectoderm and Sostdc1 to the proximal ectoderm and the mesenchyme. While Sostdc1 –/– mice lack any obvious limb or skeletal defects, Sost –/– mice recapitulate the hand defects described for Sclerosteosis patients. However, elevated WNT signaling in Sost –/–; Sostdc1 –/– mice causes misregulation of SHH signaling, ectopic activation of Sox9 in the digit 1 field and preaxial polydactyly in a Gli1- and Gli3-dependent manner. In addition, we show that the syndactyly documented in Sclerosteosis is present in both Sost –/– and Sost –/–; Sostdc1 –/– mice, and is driven by misregulation of Fgf8 in the AER, a region lacking Sost and Sostdc1 expression. This study highlights the complexity of WNT signaling in skeletalmore »
- Authors:
-
- Lawrence Livermore National Lab. (LLNL), Livermore, CA (United States). Biology and Biotechnology Division
- Lawrence Livermore National Lab. (LLNL), Livermore, CA (United States). Biology and Biotechnology Division; Univ. of California, Merced, CA (United States). School of Natural Sciences
- National Library of Medicine (NLM), Bethesda, MD (United States). National Center for Biotechnology Information. Computational Biology Branch; Univ. of Rostock (Germany). Inst. for Biostatistics and Informatics in Medicine and Ageing Research
- Regeneron Pharmaceuticals, Tarrytown, NY (United States)
- Univ. of California, Berkeley, CA (United States). Molecular and Cell Biology Dept.
- Publication Date:
- Research Org.:
- Lawrence Livermore National Laboratory (LLNL), Livermore, CA (United States)
- Sponsoring Org.:
- USDOE Office of Science (SC), Biological and Environmental Research (BER). Biological Systems Science Division
- OSTI Identifier:
- 1623726
- Grant/Contract Number:
- AC52-07NA27344
- Resource Type:
- Accepted Manuscript
- Journal Name:
- Developmental Biology
- Additional Journal Information:
- Journal Volume: 383; Journal Issue: 1; Journal ID: ISSN 0012-1606
- Publisher:
- Elsevier
- Country of Publication:
- United States
- Language:
- English
- Subject:
- 59 BASIC BIOLOGICAL SCIENCES; 97 MATHEMATICS AND COMPUTING; Developmental Biology; WNT signaling; Sost; Sclerostin; Sostdc1; Shh; Limb formation; Polydactyly; syndactyly
Citation Formats
Collette, Nicole M., Yee, Cristal S., Murugesh, Deepa, Sebastian, Aimy, Taher, Leila, Gale, Nicholas W., Economides, Aris N., Harland, Richard M., and Loots, Gabriela G. Sost and its paralog Sostdc1 coordinate digit number in a Gli3-dependent manner. United States: N. p., 2013.
Web. doi:10.1016/j.ydbio.2013.08.015.
Collette, Nicole M., Yee, Cristal S., Murugesh, Deepa, Sebastian, Aimy, Taher, Leila, Gale, Nicholas W., Economides, Aris N., Harland, Richard M., & Loots, Gabriela G. Sost and its paralog Sostdc1 coordinate digit number in a Gli3-dependent manner. United States. https://doi.org/10.1016/j.ydbio.2013.08.015
Collette, Nicole M., Yee, Cristal S., Murugesh, Deepa, Sebastian, Aimy, Taher, Leila, Gale, Nicholas W., Economides, Aris N., Harland, Richard M., and Loots, Gabriela G. Fri .
"Sost and its paralog Sostdc1 coordinate digit number in a Gli3-dependent manner". United States. https://doi.org/10.1016/j.ydbio.2013.08.015. https://www.osti.gov/servlets/purl/1623726.
@article{osti_1623726,
title = {Sost and its paralog Sostdc1 coordinate digit number in a Gli3-dependent manner},
author = {Collette, Nicole M. and Yee, Cristal S. and Murugesh, Deepa and Sebastian, Aimy and Taher, Leila and Gale, Nicholas W. and Economides, Aris N. and Harland, Richard M. and Loots, Gabriela G.},
abstractNote = {WNT signaling is critical in most aspects of skeletal development and homeostasis, and antagonists of WNT signaling are emerging as key regulatory proteins with great promise as therapeutic agents for bone disorders. Here we show that Sost and its paralog Sostdc1 emerged through ancestral genome duplication and their expression patterns have diverged to delineate nonoverlapping domains in most organ systems including musculoskeletal, cardiovascular, nervous, digestive, reproductive and respiratory. In the developing limb, Sost and Sostdc1 display dynamic expression patterns with Sost being restricted to the distal ectoderm and Sostdc1 to the proximal ectoderm and the mesenchyme. While Sostdc1 –/– mice lack any obvious limb or skeletal defects, Sost –/– mice recapitulate the hand defects described for Sclerosteosis patients. However, elevated WNT signaling in Sost –/–; Sostdc1 –/– mice causes misregulation of SHH signaling, ectopic activation of Sox9 in the digit 1 field and preaxial polydactyly in a Gli1- and Gli3-dependent manner. In addition, we show that the syndactyly documented in Sclerosteosis is present in both Sost –/– and Sost –/–; Sostdc1 –/– mice, and is driven by misregulation of Fgf8 in the AER, a region lacking Sost and Sostdc1 expression. This study highlights the complexity of WNT signaling in skeletal biology and disease and emphasizes how redundant mechanism and non-cell autonomous effects can synergize to unveil new intricate phenotypes caused by elevated WNT signaling.},
doi = {10.1016/j.ydbio.2013.08.015},
journal = {Developmental Biology},
number = 1,
volume = 383,
place = {United States},
year = {Fri Nov 01 00:00:00 EDT 2013},
month = {Fri Nov 01 00:00:00 EDT 2013}
}
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