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首页> 外文期刊>Journal of bone and mineral research: the official journal of the American Society for Bone and Mineral Research >An instrument to measure skeletal burden and predict functional outcome in fibrous dysplasia of bone.
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An instrument to measure skeletal burden and predict functional outcome in fibrous dysplasia of bone.

机译:一种测量骨骼负担并预测骨纤维发育不良的功能结局的仪器。

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摘要

An instrument to measure skeletal burden in fibrous dysplasia was developed. Biological and clinical relevance was shown by correlating skeletal burden scores with bone markers, quality of life, and ambulatory status. Childhood scores predict adult ambulatory status, and scores were unaffected when bone markers decreased with bisphosphonate treatment or aging. INTRODUCTION: Fibrous dysplasia (FD) is a skeletal disease with a broad clinical expression. There is no objective method to assess the extent of skeletal involvement or predict outcome. We developed an instrument to measure skeletal burden that correlates with physical function, health-related quality of life (HRQL), and ambulatory status. MATERIALS AND METHODS: Seventy-nine patients with FD underwent bone scintigraphy. The skeletal burden score was derived from a weighted score based on the regional measurement using bone scintigraphy to estimate the amount of FD in anatomical segments. Six readers scored 20 scans twice to determine the inter- and intrareader agreement. To assess biological significance, scores were correlated with bone markers. To assess functional outcome, scores on the SF-36 (adults) or CHQ-PF50 (children) were correlated with skeletal burden scores. In a group of patients who had bone scans as children and adults (n = 6), the ability to predict ambulatory status was tested. Skeletal burden scores were assessed in patients before and after treatment with pamidronate (n = 5). RESULTS: The inter- and intrareader agreement of burden scores were r = 0.96, and 0.98, respectively (p < 0.001 for both). The scores correlated with markers of bone metabolism and HRQL (Spearman rho, 0.54-0.67 p < 0.001 and -0.43, p = 0.001, respectively). The mean score of patients who ambulated unassisted was significantly lower than those requiring assistance (p < 0.001 unassisted versus crutch and/or wheelchair). In unassisted ambulators, younger patients had higher scores, suggesting high childhood scores may predict adulthood impairment. In six patients with childhood and adulthood scans, childhood scores >30 predicted assisted ambulation in adulthood. There was a negative correlation between bone markers and age (Spearman rho, -0.42 to -0.70; p < 0.001), but not age and skeletal burden score. Pamidronate treatment decreased serum alkaline phosphatase but had no effect on the skeletal burden score. CONCLUSIONS: This is a validated and reliable instrument for the measurement of skeletal burden of FD and is able to predict functional outcome.
机译:开发了一种测量纤维异常增生中骨骼负担的仪器。通过将骨骼负担评分与骨标志物,生活质量和非卧床状态相关联,显示出生物学和临床相关性。童年分数可预测成人的卧床状态,并且当双膦酸盐治疗或衰老导致骨标志物减少时,分数不会受到影响。简介:纤维异常增生(FD)是一种具有广泛临床表达的骨骼疾病。没有客观的方法可以评估骨骼受累程度或预测结果。我们开发了一种测量骨骼负荷的工具,该骨骼负荷与身体功能,健康相关的生活质量(HRQL)和非卧床状态相关。材料与方法:79例FD患者接受了骨闪烁显像。骨骼负担分值是根据使用骨骼闪烁显像术估计解剖段中FD量的区域测量结果得出的加权分值得出的。六名读者两次对20次扫描打分,以确定读者之间和读者内部的一致性。为了评估生物学意义,将分数与骨标记物相关联。为了评估功能结局,将SF-36(成人)或CHQ-PF50(儿童)的评分与骨骼负担评分相关。在儿童和成人(n = 6)中进行过骨扫描的一组患者中,测试了预测门诊状态的能力。在接受帕米膦酸治疗之前和之后评估骨骼负荷评分(n = 5)。结果:阅读器之间和阅读器内部的负担分值一致性分别为r = 0.96和0.98(两者均p <0.001)。得分与骨代谢和HRQL标记相关(Spearman rho,分别为0.54-0.67 p <0.001和-0.43,p = 0.001)。无助走动的患者的平均评分显着低于需要协助的患者(无助与拐杖和/或轮椅的p <0.001)。在无助走动者中,年轻的患者得分较高,这表明儿童得分较高可能预示了成年障碍。在六名接受儿童期和成年期扫描的患者中,儿童期得分> 30预计会在成年期辅助走动。骨标志物与年龄之间呈负相关(Spearman rho,-0.42至-0.70; p <0.001),但与年龄和骨骼负荷评分无相关性。帕米膦酸治疗可降低血清碱性磷酸酶,但对骨骼负荷评分无影响。结论:这是一种用于测量FD骨骼负担的经过验证且可靠的仪器,能够预测功能结果。

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