3 years ago

Serum sclerostin levels in adults with osteogenesis imperfecta: comparison with normal individuals and response to teriparatide therapy

Eric Orwoll, Sandesh C.S. Nagamani, Ying Wang, John Sloan, Brendan Lee, Rosamund Smith, Lindsey Nicol, Jay Shapiro
Sclerostin (SOST), a glycoprotein primarily derived from osteocytes, is an important regulator of bone remodeling. Osteogenesis imperfecta [1] is a heritable disorder of bone characterized by low bone mass, bone fragility, recurrent fractures, and bone deformities. Altered SOST-mediated signaling may have a role in pathogenesis of type I collagen-related OI; however this has not been evaluated in humans. We measured serum SOST levels in adults with OI who were enrolled in a randomized, placebo-controlled clinical trial that evaluated the effects of osteoanabolic therapy with teriparatide. Compared to age- and sex- matched control participants, mean SOST levels were lower in those with Type I or Types III/VI OI (P < 0.0001). Receiver operating curve analysis revealed that sclerostin alone or sclerostin plus bone mineral content discriminated patients with OI from controls (area under the curve 0.80 and 0.87 respectively). SOST levels increased in the group of patients with type I OI during therapy with teriparatide (compared to placebo, P = 0.01). The increase was significant at 6, 12, and 24 months of therapy (P < 0.02), and was apparent as early as 3 months (P = 0.06). The magnitude of increases in SOST levels during therapy was inversely correlated with increases in vertebral vBMD. Overall, these results suggest that: 1) SOST regulation is fundamentally altered in osteogenesis imperfecta, 2) serum SOST levels could be a biomarker of OI in adults, and 3) and alterations in SOST may help predict the response to anabolic therapies in OI. This article is protected by copyright. All rights reserved

Publisher URL: http://onlinelibrary.wiley.com/resolve/doi

DOI: 10.1002/jbmr.3312

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