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OI is a heterogeneous group of heritable bone disorders often caused by a mutation affecting one of two genes that encode collagen type 1 chains. It is characterized by low bone density and bone fragility, with gender-related differences.Current pharmaceutical treatment (bisphosphonates) has undesirable effects. By its osteogenic effect, sclerostin antibody (Scl-Ab) has recently emerged as a potential treatment of OI. We used Oim-/- mice, a validated model of human type 3 OI, to study gender-related impact of Scl-Ab. Normal (Wt) and Oim mice aged 4 weeks received weekly intraperitoneal injections of vehicle (Veh) or Scl-Ab during 9 weeks. The effects of Scl-Ab on clinimetric data and quantitative bone parameters in male and female tibiae were studied using X-rays, pQCT, microradiography and dynamic histomorphometry (after in vivo fluorescent labelling). Contrary to Wt mice, female Oim were longer than males and most of their bone parameters were higher at 13 weeks, though the number of fractures was higher in male than female Oim. Scl-Ab treatment increased bone mineral density and histomorphometric bone parameters in Wt and Oim mice. These increases allowed male Oim to reach similar values than females. However, Oim mice remained smaller than Wt.