Sex-specific trisomic Dyrk1a-related skeletal phenotypes during development in a Down syndrome model.

ABSTRACT: Skeletal insufficiency affects all individuals with Down syndrome (DS) or trisomy 21 and may alter bone strength throughout development due to a reduced period of bone formation and early attainment of peak bone mass compared to those in typically developing individuals. Appendicular skeletal deficits also appear in males before females with DS. In femurs of male Ts65Dn DS model mice, cortical deficits were pronounced throughout development, but trabecular deficits and Dyrk1a overexpression were transitory until postnatal day (P) 30, when there were persistent trabecular and cortical deficits and Dyrk1a was trending toward overexpression. Correction of DS-related skeletal deficits by a purported DYRK1A inhibitor or through genetic means beginning at P21 was not effective at P30, but germli

SUBMITTER: LaCombe JM 

PROVIDER: S-EPMC11449447 | biostudies-literature | 2024 Sep

REPOSITORIES: biostudies-literature