Small molecule inhibitors of the Dishevelled-CXXC5 interaction are new drug candidates for bone anabolic osteoporosis therapy

EMBO Mol Med. 2016 Apr 1;8(4):375-87. doi: 10.15252/emmm.201505714.

Abstract

Bone anabolic agents promoting bone formation and rebuilding damaged bones would ideally overcome the limitations of anti-resorptive therapy, the current standard prescription for osteoporosis. However, the currently prescribed parathyroid hormone (PTH)-based anabolic drugs present limitations and adverse effects including osteosarcoma during long-term use. Also, the antibody-based anabolic drugs that are currently being developed present the potential limits in clinical application typical of macromolecule drugs. We previously identified that CXXC5 is a negative feedback regulator of the Wnt/β-catenin pathway via its interaction with Dishevelled (Dvl) and suggested the Dvl-CXXC5 interaction as a potential target for anabolic therapy of osteoporosis. Here, we screened small-molecule inhibitors of the Dvl-CXXC5 interaction via a newly established in vitro assay system. The screened compounds were found to activate the Wnt/β-catenin pathway and enhance osteoblast differentiation in primary osteoblasts. The bone anabolic effects of the compounds were shown using ex vivo-cultured calvaria. Nuclear magnetic resonance (NMR) titration analysis confirmed interaction between Dvl PDZ domain and KY-02061, a representative of the screened compounds. Oral administration of KY-02327, one of 55 newly synthesized KY-02061 analogs, successfully rescued bone loss in the ovariectomized (OVX) mouse model. In conclusion, small-molecule inhibitors of the Dvl-CXXC5 interaction that block negative feedback regulation of Wnt/β-catenin signaling are potential candidates for the development of bone anabolic anti-osteoporosis drugs.

Keywords: CXXC5; Dishevelled; Wnt/β‐catenin pathway; negative feedback regulation; osteoporosis.

MeSH terms

  • Administration, Oral
  • Animals
  • DNA-Binding Proteins
  • Dishevelled Proteins / antagonists & inhibitors*
  • Dishevelled Proteins / metabolism*
  • Drug Evaluation, Preclinical / methods
  • Intracellular Signaling Peptides and Proteins / antagonists & inhibitors*
  • Intracellular Signaling Peptides and Proteins / metabolism*
  • Mice
  • Organ Culture Techniques
  • Osteoblasts / drug effects
  • Osteoporosis / drug therapy*
  • Protein Binding / drug effects
  • Skull / drug effects
  • Skull / growth & development
  • Transcription Factors
  • Treatment Outcome
  • Wnt Signaling Pathway / drug effects

Substances

  • CXXC5 protein, mouse
  • DNA-Binding Proteins
  • Dishevelled Proteins
  • Intracellular Signaling Peptides and Proteins
  • Transcription Factors