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Teriparatide Treatment in Adult Patients with Osteogenesis Imperfecta Type I

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Abstract

Osteogenesis imperfecta (OI) is a hereditary disease characterized by low bone mass, increased bone fragility, short stature, and skeletal deformities. This study focuses on OI type I, the mildest form of the disease. Bisphosphonates represent the prevailing standard of care in patients with OI. Teriparatide (TPD) is a PTH analog with bone-anabolic actions which has been approved for osteoporosis treatment. Thirteen postmenopausal women with type I OI who had been on treatment with neridronate for at least 2 years and who incurred new vertebral fracture during treatment were treated with TPD for 18 months. Bone mineral density (BMD) increased significantly over 18 months up to 3.5 % at the lumbar spine (p = 0.001), while no significant changes were noted in hip BMD. Serum markers of bone formation and of bone resorption increased significantly during the treatment. The Wnt inhibitors serum dickkopf-1 (DKK1) and sclerostin were also measured. A nonsignificant increase was seen in serum sclerostin levels, while serum DKK1 rose gradually and significantly during TPD treatment. In patients affected by type I OI, TPD treatment is associated with a remarkable response in markers of bone formation. This suggests a normal osteoblastic response to TPD. However, the observed increases in BMD were somewhat lower than those in postmenopausal or senile osteoporosis treated with TPD for the same lag time. Our results open the possibility to develop TPD for the treatment of adult type I OI, but particularly for the lack of a control group, a properly designed controlled study is warranted.

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Acknowledgments

We thank Caterina Fraccarollo and Cristina Bosco for the ELISAs.

Conflict of interest

S. Adami has consultant/advisory role to Amgen and Merck. All other authors have stated that they have no conflict of interest.

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Authors and Affiliations

  1. Rheumatology Unit, Department of Medicine, University of Verona, Piazzale Stefani 1, 37126, Verona, Italy

    Davide Gatti, Maurizio Rossini, Ombretta Viapiana, Maria Rosaria Povino, Saverio Liuzza, Elena Fracassi, Luca Idolazzi & Silvano Adami

Authors
  1. Davide Gatti
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  2. Maurizio Rossini
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  3. Ombretta Viapiana
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  4. Maria Rosaria Povino
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  5. Saverio Liuzza
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  6. Elena Fracassi
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  7. Luca Idolazzi
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  8. Silvano Adami
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Correspondence to Davide Gatti.

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Gatti, D., Rossini, M., Viapiana, O. et al. Teriparatide Treatment in Adult Patients with Osteogenesis Imperfecta Type I. Calcif Tissue Int 93, 448–452 (2013). https://doi.org/10.1007/s00223-013-9770-2

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  • DOI: https://doi.org/10.1007/s00223-013-9770-2

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