Male Down syndrome Ts65Dn mice have impaired bone regeneration

Bone. 2024 Dec 13:192:117374. doi: 10.1016/j.bone.2024.117374. Online ahead of print.

Abstract

Trisomy of human chromosome 21 (Ts21) individuals present with a spectrum of low bone mineral density (BMD) that predisposes this vulnerable group to skeletal injuries. To determine the bone regenerative capacity of Down syndrome (DS) mice, male and female Dp16 and Ts65Dn DS mice underwent amputation of the digit tip (the terminal phalanx (P3)). This is a well-established mammalian model of bone regeneration that restores the amputated skeletal segment and all associated soft tissues. P3 amputation was performed in 8-week-old male and female DS mice and WT controls and followed by in vivo μCT, histology and immunofluorescence. Following P3 amputation, the bone degradation phase was attenuated in both Dp16 and Ts65Dn males. In Dp16 males, P3 regeneration was delayed but complete by 63 days post amputation (DPA); however, male Ts65Dn exhibited attenuated regeneration by 63 DPA. In both Dp16 and Ts65Dn female DS mice, P3 regenerates were indistinguishable from WT by 42 DPA. In Ts65Dn males, osteoclasts and eroded bone surface were significantly reduced, and osteoblast number significantly decreased in the regenerating digit. In Ts65Dn females, no significant differences were observed in any osteoclast or osteoblast parameter. Like Ts21 individuals and DS mice with sex differences in bone mass, these data expand the characteristic sexually dimorphism to include bone resorption and regeneration in response to skeletal injury in Ts65Dn mice. These observations suggest that sex differences contribute to the poor bone healing of DS and compound the increased risk of bone injury in the Ts21 population.

Keywords: Bone; Digit; Down syndrome; Dp16; P3; Regeneration; Ts65Dn.