Disruption of hedgehog signaling leads to hyoid bone dysplasia during embryogenesis

Differentiation. 2023 May-Jun:131:82-88. doi: 10.1016/j.diff.2023.05.001. Epub 2023 May 8.

Abstract

The development of the hyoid bone is a complex process that involves the coordination of multiple signaling pathways. Previous studies have demonstrated that disruption of the hedgehog pathway in mice results in a series of structural malformations. However, the specific role and critical period of the hedgehog pathway in the early development of the hyoid bone have not been thoroughly characterized. In this study, we treated pregnant ICR mice with the hedgehog pathway inhibitor vismodegib by oral gavage in order to establish a model of hyoid bone dysplasia. Our results indicate that administration of vismodegib at embryonic days 11.5 (E11.5) and E12.5 resulted in the development of hyoid bone dysplasia. We were able to define the critical periods for the induction of hyoid bone deformity through the use of a meticulous temporal resolution. Our findings suggest that the hedgehog pathway plays a crucial role in the early development of the hyoid bone. Additionally, our research has established a novel and easily established mouse model of synostosis in the hyoid bone using a commercially available pathway-selective inhibitor.

Keywords: Cartilage; Development; Hedgehog signaling; Hyoid bone.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Animals
  • Bone Diseases, Developmental*
  • Embryonic Development
  • Female
  • Hedgehog Proteins* / genetics
  • Hedgehog Proteins* / metabolism
  • Hyoid Bone / metabolism
  • Mice
  • Mice, Inbred ICR
  • Pregnancy

Substances

  • Hedgehog Proteins
  • HhAntag691