HEMATIM UR-UPJV 4666, C.U.R.S, University of Picardie Jules Verne, 80000 Amiens, France; Centre of Molecular Inflammation Research (CEMIR), Department of Clinical Research and Molecular Medicine (IKOM), Faculty of Medicine and Health Sciences (MH), Norwegian University of Science and Technology (NTNU), Trondheim, Norway.
INSERM UMRS 1148, Laboratory for Vascular Translational Science (LVTS), UFR SMBH, University of Sorbonne Paris Nord, 93000 Bobigny, France.
Bone. 2024 Oct;187:117205. doi: 10.1016/j.bone.2024.117205. Epub 2024 Jul 15.
MULIBREY nanism which results from autosomal recessive mutations in TRIM37 impacts skeletal development, leading to growth delay with complications in multiple organs. In this study, we employed a combined proteomics and qPCR screening approach to investigate the molecular alterations in the CHON-002 cell line by comparing CHON-002 wild-type (WT) cells to CHON-002 TRIM37 knockdown (KD) cells. Our proteomic analysis demonstrated that TRIM37 depletion predominantly affects the expression of extracellular matrix proteins (ECM). Specifically, nanoLC-MS/MS experiments revealed an upregulation of SPARC, and collagen products (COL1A1, COL3A1, COL5A1) in response to TRIM37 KD. Concurrently, large-scale qPCR assays targeting osteogenesis-related genes corroborated these dysregulations of SPARC at the mRNA level. Gene ontology enrichment analysis highlighted the involvement of dysregulated proteins in ECM organization and TGF-β signaling pathways, indicating a role for TRIM37 in maintaining ECM integrity and regulating chondrocyte proliferation. These findings suggest that TRIM37 deficiency in chondrocytes change ECM protein composition and could impairs long bone growth, contributing to the pathophysiology of MULIBREY nanism.
MULIBREY 矮小症是由 TRIM37 的常染色体隐性突变引起的,影响骨骼发育,导致生长迟缓,并伴有多个器官的并发症。在这项研究中,我们采用了蛋白质组学和 qPCR 联合筛选方法,通过比较 CHON-002 野生型(WT)细胞和 CHON-002 TRIM37 敲低(KD)细胞,研究 CHON-002 细胞系中的分子变化。我们的蛋白质组学分析表明,TRIM37 耗竭主要影响细胞外基质蛋白(ECM)的表达。具体来说,nanoLC-MS/MS 实验显示,TRIM37 KD 后 SPARC 和胶原蛋白产物(COL1A1、COL3A1、COL5A1)表达上调。同时,针对成骨相关基因的大规模 qPCR 检测证实了 SPARC 在 mRNA 水平的这些失调。基因本体富集分析强调了失调蛋白在细胞外基质组织和 TGF-β信号通路中的作用,表明 TRIM37 在维持细胞外基质完整性和调节软骨细胞增殖方面发挥作用。这些发现表明,软骨细胞中 TRIM37 的缺乏改变了 ECM 蛋白组成,并可能损害长骨生长,导致 MULIBREY 矮小症的病理生理学改变。