USF3 modulates osteoporosis risk by targeting WNT16, RANKL, RUNX2, and two GWAS lead SNPs rs2908007 and rs4531631

Hum Mutat. 2021 Jan;42(1):37-49. doi: 10.1002/humu.24126. Epub 2020 Nov 30.

Abstract

Osteoporotic fractures cause major morbidity and mortality in the aging population. Genome-wide association studies (GWAS) have identified USF3 as the novel susceptibility gene of osteoporosis. However, the functional role in bone metabolism and the target gene of the basic helix-loop-helix transcription factor USF3 are unclear. Here, we show that USF3 enhances osteoblast differentiation and suppresses osteoclastogenesis in cultured human osteoblast-like U-2OS cells. Mechanistic studies revealed that transcription factor USF3 antagonistically interacts with anti-osteogenic TWIST1/TCF12 heterodimer in the WNT16 and RUNX2 promoter, and counteracts CREB1 and JUN/FOS in the RANKL promoter. Importantly, the osteoporosis GWAS variant g.1744A>G (rs2908007A>G) located in the WNT16 promoter confers G-allele-specific transcriptional modulation by USF3, TWIST1/TCF12 and TBX5/TBX15, and USF3 transactivates the osteoclastogenesis suppressor WNT16 promoter activity and antagonizes the repression of WNT16 by TWIST1 and TCF12. The risk G allele of osteoporosis GWAS variant g.3260A>G (rs4531631A>G) in the RANKL promoter facilitates the binding of CREB1 and JUN/FOS and enhances transactivation of the osteoclastogenesis contributor RANKL that is inhibited by USF3. Our findings uncovered the functional mechanisms of osteoporosis novel GWAS-associated gene USF3 and lead single nucleotide polymorphisms rs2908007 and rs4531631 in the regulation of bone formation and resorption.

Keywords: GWAS SNP; RANKL; RUNX2; USF3; WNT16; osteoporosis.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Aged
  • Basic Helix-Loop-Helix Transcription Factors / genetics*
  • Cell Differentiation / genetics
  • Core Binding Factor Alpha 1 Subunit / genetics
  • Core Binding Factor Alpha 1 Subunit / metabolism
  • Genome-Wide Association Study*
  • Humans
  • Osteoblasts
  • Osteoporosis* / genetics
  • Polymorphism, Single Nucleotide
  • RANK Ligand / genetics
  • T-Box Domain Proteins / genetics
  • Wnt Proteins / genetics
  • Wnt Proteins / metabolism

Substances

  • Basic Helix-Loop-Helix Transcription Factors
  • Core Binding Factor Alpha 1 Subunit
  • RANK Ligand
  • RUNX2 protein, human
  • T-Box Domain Proteins
  • TBX15 protein, human
  • TNFSF11 protein, human
  • USF3 protein, human
  • WNT16 protein, human
  • Wnt Proteins