Department of Immunology and Infectious Disease, Harvard School of Public Health, Boston, MA 02115, USA.
Proc Natl Acad Sci U S A. 2012 May 22;109(21):8173-8. doi: 10.1073/pnas.1205848109. Epub 2012 May 9.
Mice lacking the large zinc finger protein Schnurri-3 (Shn3) display increased bone mass, in part, attributable to augmented osteoblastic bone formation. Here, we show that in addition to regulating bone formation, Shn3 indirectly controls bone resorption by osteoclasts in vivo. Although Shn3 plays no cell-intrinsic role in osteoclasts, Shn3-deficient animals show decreased serum markers of bone turnover. Mesenchymal cells lacking Shn3 are defective in promoting osteoclastogenesis in response to selective stimuli, likely attributable to reduced expression of the key osteoclastogenic factor receptor activator of nuclear factor-κB ligand. The bone phenotype of Shn3-deficient mice becomes more pronounced with age, and mice lacking Shn3 are completely resistant to disuse osteopenia, a process that requires functional osteoclasts. Finally, selective deletion of Shn3 in the mesenchymal lineage recapitulates the high bone mass phenotype of global Shn3 KO mice, including reduced osteoclastic bone catabolism in vivo, indicating that Shn3 expression in mesenchymal cells directly controls osteoblastic bone formation and indirectly regulates osteoclastic bone resorption.
缺乏大锌指蛋白 Schnurri-3 (Shn3) 的小鼠表现出骨量增加,部分原因可归因于成骨细胞骨形成的增加。在这里,我们表明 Shn3 除了调节骨形成外,还在体内通过破骨细胞间接控制骨吸收。尽管 Shn3 在破骨细胞中没有发挥细胞内在的作用,但 Shn3 缺陷动物表现出降低的血清骨转换标志物。缺乏 Shn3 的间充质细胞在响应选择性刺激时促进破骨细胞生成的能力受损,可能归因于关键破骨细胞生成因子核因子-κB 配体激活物受体的表达减少。Shn3 缺陷小鼠的骨表型随着年龄的增长变得更加明显,并且 Shn3 缺陷小鼠完全抵抗废用性骨质疏松症,这一过程需要功能性破骨细胞。最后,间充质谱系中 Shn3 的选择性缺失重现了全局 Shn3 KO 小鼠的高骨量表型,包括体内破骨细胞骨代谢减少,表明间充质细胞中 Shn3 的表达直接控制成骨细胞骨形成,并间接调节破骨细胞骨吸收。