Kitazawa Riko, Haraguchi Ryuma, Kitazawa Sohei
Department of Molecular Pathology, Ehime University Graduate School of Medicine, 454 Shitsukawa, Toon-City, Ehime, 791-0295, Japan.
Sci Rep. 2025 Jul 31;15(1):27919. doi: 10.1038/s41598-025-13314-7.
Bone remodeling is tightly regulated by the RANK-RANKL-OPG axis to maintain skeletal integrity. We previously identified a RANK splicing variant, vRANK, which encodes a truncated protein that inhibits osteoclastogenesis and promotes apoptosis. This study examined the regulatory mechanisms of vRANK expression in vitro and its physiological role in vivo using transgenic mice with forced vRANK expression in the monocyte-macrophage lineage and in a systemic manner. In vitro, PMA and TGF-β1 specifically induced vRANK expression, which was completely suppressed by U0126, a MEK1/2 inhibitor, and significantly reduced by Sam68 knockdown, indicating its involvement in RNA splicing regulation. In vivo, RANK-Cre-driven vRANK expression had no skeletal impact, whereas LysM-Cre-driven expression resulted in increased bone mass and suppressed osteoclastogenesis. However, systemic overexpression of vRANK (CAG-CreER) caused perinatal lethality, severe cardiac fibrosis, and immune dysfunction. Notably, myocardial fibrosis in vRANK-overexpressing mice correlated with TGF-β1 upregulation in fibrotic foci, suggesting a pathological feedback loop exacerbating fibrosis. These findings suggest that vRANK functions similarly to OPG in inhibiting osteoclastogenesis while also potentially playing a role in cardiac remodeling and immune regulation.
骨重塑受RANK-RANKL-OPG轴严格调控以维持骨骼完整性。我们之前鉴定出一种RANK剪接变体vRANK,它编码一种截短的蛋白,可抑制破骨细胞生成并促进细胞凋亡。本研究使用在单核细胞-巨噬细胞谱系中强制表达vRANK的转基因小鼠,以全身方式研究了vRANK在体外的调控机制及其在体内的生理作用。在体外,PMA和TGF-β1特异性诱导vRANK表达,MEK1/2抑制剂U0126可完全抑制该表达,而Sam68敲低可显著降低该表达,表明其参与RNA剪接调控。在体内,RANK-Cre驱动的vRANK表达对骨骼无影响,而LysM-Cre驱动的表达导致骨量增加并抑制破骨细胞生成。然而,vRANK的全身过表达(CAG-CreER)导致围产期死亡、严重的心脏纤维化和免疫功能障碍。值得注意的是,vRANK过表达小鼠的心肌纤维化与纤维化病灶中TGF-β1上调相关,提示存在加剧纤维化的病理反馈环。这些发现表明,vRANK在抑制破骨细胞生成方面的功能与OPG相似,同时也可能在心脏重塑和免疫调节中发挥作用。