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Serum Sclerostin Levels in Adults With Osteogenesis Imperfecta: Comparison With Normal Individuals and Response to Teriparatide Therapy.

  • Nicol, Lindsey1
  • Wang, Ying2
  • Smith, Rosamund3
  • Sloan, John3
  • Nagamani, Sandesh Cs4, 5
  • Shapiro, Jay6
  • Lee, Brendan4, 5
  • Orwoll, Eric7
  • 1 Department of Pediatrics, Division of Endocrinology, Oregon Health & Sciences University, Portland, OR, USA.
  • 2 Department of Medicine, Division of Biostatistics, Oregon Health & Sciences University, Portland, OR, USA.
  • 3 Lilly Research Laboratories, Indianapolis, IN, USA. , (India)
  • 4 Department of Molecular and Human Genetics, Baylor College of Medicine, Houston, TX, USA.
  • 5 Texas Children's Hospital, Houston, TX, USA.
  • 6 Department of Bone and Osteogenesis Imperfecta, Kennedy Krieger Institute, Baltimore, MD, USA.
  • 7 Department of Medicine, Division of Endocrinology, Oregon Health & Sciences University, Portland, OR, USA.
Published Article
Journal of Bone and Mineral Research
Wiley (John Wiley & Sons)
Publication Date
Feb 01, 2018
DOI: 10.1002/jbmr.3312
PMID: 29044725


Sclerostin (SOST), a glycoprotein primarily derived from osteocytes, is an important regulator of bone remodeling. Osteogenesis imperfecta (OI) 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 with 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 with 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 volumetric bone mineral density (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) alterations in SOST may help predict the response to anabolic therapies in OI. © 2017 American Society for Bone and Mineral Research. © 2017 American Society for Bone and Mineral Research.

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