Growth characteristics in individuals with osteogenesis imperfecta in North America: results from a multicenter study.
Growth characteristics in individuals with osteogenesis imperfecta in North America: results from a multicenter study.
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DOI：
10.1038/s41436-018-0045-1
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发表时间：
2019-03
期刊：
影响因子：
--
通讯作者：
Nagamani SCS
中科院分区：
文献类型：
--
作者：
Jain M;Tam A;Shapiro JR;Steiner RD;Smith PA;Bober MB;Hart T;Cuthbertson D;Krischer J;Mullins M;Bellur S;Byers PH;Pepin M;Durigova M;Glorieux FH;Rauch F;Lee B;Sutton VR;, Members of the Brittle Bone Disorders Consortium*,;Nagamani SCS
成骨不全（OI）易导致复发性骨折、骨畸形和身材矮小。有一个缺乏大规模的系统研究，调查生长参数在OI。使用来自连锁临床研究中心的数据，我们比较了552名OI患者的身高、生长速度、体重和体重指数（BMI）。将身高、体重和BMI绘制在疾病控制中心的标准曲线上。在儿童中，I型、III型和IV型OI的身高中位Z评分分别为-0.66、-6.91和-2.79。III型和IV型OI的生长速度降低。III型OI儿童的体重中位Z评分为-4.55。I型、III型和IV型OI儿童的BMI中位Z评分分别为0.10、0.91和0.67。广义线性模型分析表明，身高Z评分与OI亚型的严重程度（p<0.001）、年龄、双膦酸盐使用和使用棒（p<0.05）呈正相关。从最大的OI患者队列中，我们提供了身高、体重和BMI Z评分的中位数，这可以帮助评估诊所环境中的总体生长情况。这项研究是生成OI特异性生长曲线的重要的第一步。
Osteogenesis imperfecta (OI) predisposes to recurrent fractures, bone deformities, and short stature. There is a lack of large-scale systematic studies that have investigated growth parameters in OI. Using data from the Linked Clinical Research Centers, we compared height, growth velocity, weight, and body mass index (BMI) in 552 individuals with OI. Height, weight, and BMI were plotted on the Centers for Disease Control normative curves. In children, the median Z-scores for height in OI types I, III, and IV were −0.66, −6.91, and −2.79, respectively. Growth velocity was diminished in OI types III and IV. The median Z-score for weight in children with OI type III was −4.55. The median Z-scores for BMI in children with OI types I, III and IV were 0.10, 0.91 and 0.67, respectively. Generalized linear model analyses demonstrated height Z-score positively correlated with severity of OI subtype (p<0.001), age, bisphosphonate use, and rodding (p<0.05). From the largest cohort of individuals with OI, we provide median values for height, weight, and BMI Z-scores which can aid in evaluation of overall growth in the clinic setting. This study is an important first step in the generation of OI-specific growth curves.
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影响因子：
5.2
作者：
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通讯作者：
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作者：
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