Dental tissue changes in juvenile and adult mice with osteogenesis imperfecta

Anat Rec (Hoboken). 2024 Mar;307(3):600-610. doi: 10.1002/ar.25306. Epub 2023 Aug 28.

Abstract

Osteogenesis imperfecta (OI), a disorder of type I collagen, causes skeletal deformities as well as defects in dental tissues, which lead to increased enamel wear and smaller teeth with shorter roots. Mice with OI exhibit similar microstructural dentin changes, including reduced dentin tubule density and dentin cross-sectional area. However, the effects of these mutations on gross dental morphology and dental tissue volumes have never been characterized in the osteogenesis imperfecta murine (OIM) mouse model. Here we compare mineralized dental tissue measurements of OIM mice and unaffected wild type (WT) littermates at the juvenile and adult stages. The maxillary and mandibular incisors and first molars were isolated from microCT scans, and tissue volumes and root length were measured. OIM mice have smaller teeth with shorter roots relative to WT controls. Maxillary incisor volumes differed significantly between OIM and WT mice at both the juvenile and young adult stage, perhaps due to shortening of the maxilla itself in OIM mice. Additionally, adult OIM mice have significantly less crown enamel volume than do juveniles, potentially due to loss through wear. Thus, OIM mice demonstrate a dental phenotype similar to humans with OI, with decreased tooth size, decreased root length, and accelerated enamel wear. Further investigation of dental development in the OIM mouse may have important implications for the development and treatment of dental issues in OI patients.

Keywords: COL1A; DI; OIM; dentin; dentinogenesis imperfecta; microCT; odontogenesis.

MeSH terms

  • Animals
  • Collagen Type I
  • Disease Models, Animal
  • Humans
  • Incisor
  • Mice
  • Mutation
  • Osteogenesis Imperfecta* / genetics
  • Phenotype

Substances

  • Collagen Type I