Versiti Blood Research Institute, Milwaukee, WI, 53213, USA.
Biomedical Research Center of South China, Fujian Normal University, Fujian, 350117, China.
Leukemia. 2023 Nov;37(11):2261-2275. doi: 10.1038/s41375-023-02014-8. Epub 2023 Sep 5.
The highly conserved MicroRNA-9 (miR-9) family consists of three members. We discovered that miR-9-1 deletion reduced mature miR-9 expression, causing 43% of the mice to display smaller size and postweaning lethality. MiR-9-1-deficient mice with growth defects experienced severe lymphopenia, but other blood cells were unaffected. The lymphopenia wasn't due to defects in hematopoietic progenitors, as mutant bone marrow (BM) cells underwent normal lymphopoiesis after transplantation into wild-type recipients. Additionally, miR-9-1-deficient mice exhibited impaired osteoblastic bone formation, as mutant mesenchymal stem cells (MSCs) failed to differentiate into osteoblastic cells (OBs). RNA sequencing revealed reduced expression of master transcription factors for osteoblastic differentiation, Runt-related transcription factor 2 (Runx2) and Osterix (Osx), and genes related to collagen formation, extracellular matrix organization, and cell adhesion, in miR-9-1-deficient MSCs. Follistatin (Fst), an antagonist of bone morphogenetic proteins (BMPs), was found to be a direct target of miR-9-1. Its deficiency led to the up-regulation of Fst, inhibiting BMP signaling in MSCs, and reducing IL-7 and IGF-1. Thus, miR-9-1 controls osteoblastic regulation of lymphopoiesis by targeting the Fst/BMP/Smad signaling axis.
高度保守的 MicroRNA-9 (miR-9) 家族由三个成员组成。我们发现 miR-9-1 的缺失会减少成熟 miR-9 的表达,导致 43%的小鼠体型变小,且断奶后死亡。具有生长缺陷的 miR-9-1 缺陷型小鼠出现严重的淋巴细胞减少症,但其他血细胞不受影响。淋巴细胞减少症不是由于造血祖细胞缺陷引起的,因为突变的骨髓 (BM) 细胞在移植到野生型受体后仍能正常进行淋巴生成。此外,miR-9-1 缺陷型小鼠表现出成骨细胞骨形成受损,因为突变的间充质干细胞 (MSC) 无法分化为成骨细胞 (OB)。RNA 测序显示,miR-9-1 缺陷型 MSC 中成骨分化的主转录因子 Runt 相关转录因子 2 (Runx2) 和骨钙素 (Osx) 以及与胶原形成、细胞外基质组织和细胞黏附相关的基因表达降低。卵泡抑素 (Fst) 是骨形态发生蛋白 (BMPs) 的拮抗剂,被发现是 miR-9-1 的直接靶点。它的缺乏导致 Fst 的上调,抑制了 MSC 中的 BMP 信号通路,并减少了 IL-7 和 IGF-1。因此,miR-9-1 通过靶向 Fst/BMP/Smad 信号轴来控制成骨细胞对淋巴生成的调节。