Combined antiresorptive and new anabolic drug approach in osteogenesis imperfecta zebrafish models.

ABSTRACT: Osteogenesis imperfecta (OI) is a family of heritable collagen I-related skeletal disorders for which, to date, no definitive cure is available. Individuals with OI are mainly treated with bisphosphonates that enhance bone mass by inhibiting bone resorption. However, new strategies combining antiresorptive molecules with bone anabolic drugs are likely to provide valid alternatives for skeletal health, protecting physiological bone turnover. Recently, cellular stress has been identified as a therapeutic target in both dominant and recessive forms of OI characterized by overmodified collagen I. The chemical chaperone 4-phenylbutyrate (4PBA) successfully ameliorated cell homeostasis in both in vitro and in vivo OI models. In this study, dominant Chihuahua (Chi/+) and recessive <

SUBMITTER: Masiero C 

PROVIDER: S-EPMC12356371 | biostudies-literature | 2025 Sep

REPOSITORIES: biostudies-literature