Bains Jaskaran S, Carter Erin M, Citron Kate P, Boskey Adele L, Shapiro Jay R, Steiner Robert D, Smith Peter A, Bober Michael B, Hart Tracy, Cuthbertson David, Krischer Jeff, Byers Peter H, Pepin Melanie, Durigova Michaela, Glorieux Francis H, Rauch Frank, Sliepka Joseph M, Sutton V Reid, Lee Brendan, Nagamani Sandesh Cs, Raggio Cathleen L
Hospital for Special Surgery Dept of Orthopedic Surgery New York NY USA.
Department of Bone and Osteogenesis Imperfecta Kennedy Krieger Institute Baltimore MD USA.
JBMR Plus. 2019 Jan 7;3(5):e10118. doi: 10.1002/jbm4.10118. eCollection 2019 May.
Osteogenesis imperfecta (OI) is characterized by low bone mass and bone fragility. Using data from a large cohort of individuals with OI from the Osteogenesis Imperfecta Foundation's linked clinical research centers, we examined the association between exposure to bisphosphonate (BPN) treatment (past or present) and lumbar spine (LS) areal bone mineral density (aBMD), fractures, scoliosis, and mobility. From 466 individuals, we obtained 1394 participant-age LS aBMD data points. Though all OI subtypes were examined, primary analyses were restricted to type I OI (OI-1). Using linear regression, we constructed expected OI-1 LS aBMD-for-age curves from the data from individuals who had never received BPN. LS aBMD in those who had been exposed to BPN was then compared with the computed expected aBMD. BPN exposure in preadolescent years (age <14 years) was associated with a LS aBMD that was 9% more than the expected computed values in BPN-naïve individuals ( < 0.01); however, such association was not observed across all ages. Exposure to i.v. BPN and treatment duration >2 years correlated with LS aBMD in preadolescent individuals. BPN exposure also had a significant association with non-aBMD clinical outcome variables. Logistic regression modeling predicted that with BPN exposure, a 1-year increase in age would be associated with an 8.2% decrease in fracture probability for preadolescent individuals with OI-1, compared with no decrease in individuals who had never received any BPN ( < 0.05). In preadolescent individuals with OI-1, a 0.1 g/cm increase in LS aBMD was associated with a 10.6% decrease in scoliosis probability, compared with a 46.8% increase in the BPN-naïve group ( < 0.01). For the same changes in age and LS aBMD in preadolescent individuals, BPN exposure was also associated with higher mobility scores ( < 0.01), demonstrating that BPN treatment may be associated with daily function. © 2018 The Authors. Published by Wiley Periodicals, Inc. on behalf of American Society for Bone and Mineral Research.
成骨不全症（OI）的特征是骨量低和骨骼脆弱。利用来自成骨不全症基金会关联临床研究中心的大量成骨不全症患者队列的数据，我们研究了双膦酸盐（BPN）治疗（过去或现在）的暴露与腰椎（LS）面积骨密度（aBMD）、骨折、脊柱侧弯和活动能力之间的关联。在466名个体中，我们获得了1394个参与者年龄对应的LS aBMD数据点。尽管对所有OI亚型都进行了检查，但主要分析仅限于I型OI（OI-1）。我们使用线性回归，根据从未接受过BPN治疗的个体的数据构建了预期的OI-1 LS年龄别aBMD曲线。然后将接受过BPN治疗的个体的LS aBMD与计算得出的预期aBMD进行比较。青春期前（年龄<14岁）接触BPN与LS aBMD比未接触过BPN的个体（<0.01）的预期计算值高9%相关；然而，并非在所有年龄段都观察到这种关联。静脉注射BPN和治疗持续时间>2年与青春期前个体的LS aBMD相关。BPN暴露与非aBMD临床结局变量也有显著关联。逻辑回归模型预测，对于接触BPN的OI-1青春期前个体，年龄每增加1岁，骨折概率将降低8.2%，而从未接受过任何BPN治疗的个体则无降低（<0.05）。在OI-1青春期前个体中，LS aBMD每增加0.1 g/cm，脊柱侧弯概率降低10.6%，而在未接触过BPN的组中则增加46.8%（<0.01）。对于青春期前个体年龄和LS aBMD的相同变化，BPN暴露还与更高的活动能力评分相关（<0.01），表明BPN治疗可能与日常功能有关。© 2018作者。由Wiley Periodicals, Inc.代表美国骨与矿物质研究学会出版。
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