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Rehberg, Mirko; Winzenrieth, Renaud; Hoyer-Kuhn, Heike; Duran, Ibrahim; Schoenau, Eckhard; Semler, Oliver

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Abstract Note

Introduction/Background Osteogenesis imperfecta is a hereditary connective tissue disorder, resulting in low bone mass and high bone fragility. Dual-energy X-ray Absorptiometry (DXA) and in adulthood also the Trabecular Bone score (TBS) are well established to assess bone health and fracture risk. The purpose of this investigation was to assess the usefulness of TBS in respect to different treatment regimes in children with Osteogenesis imperfecta (OI). Changes of areal bone mineral density (aBMD) and TBS using DXA scans of children treated with antiresorptive therapies were evaluated. Methodology DXA scans (aBMD, TBS) of 8 children with OI were evaluated. The scans were taken during a 1 year period of treatment with bisphosphonates and during one year pilot trial using denosumab. Changes of aBMD and TBS during both treatment regimens were compared. Results During bisphosphonate treatment aBMD increased about 6.2 %, while TBS increased about 2.1 %. The difference between aBMD and TBS before and after bisphosphonate treatment was not significant (p = 0.25). During denosumab treatment aBMD increased around 25.1 %, while TBS increased 6.7 %. The change of aBMD was significant (p = 0.007), as was the difference between aBMD and TBS (p < 0.001). Conclusions Denosumab had a significant effect on both aBMD and TBS but was significantly more pronounced in aBMD. These results suggest a stronger effect of denosumab on cortical bone and the growth plate in comparison to bisphosphonates. Beside the lack of paediatric reference data and the small sample size, the results suggest TBS to be a useful tool for monitoring skeletal changes during development, growth and antiresorptive therapy in children with OI.


Journal of Clinical Densitometry



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