Haeusler G, Frisch H
Paediatric Department, University of Vienna, Austria.
Acta Paediatr. 1994 Mar;83(3):309-14. doi: 10.1111/j.1651-2227.1994.tb18100.x.
Patients with Ullrich-Turner syndrome (UTS) are treated increasingly with growth hormone (GH) to improve growth velocity and final height. For evaluation of the effectiveness of this treatment, different methods have been applied and we demonstrate that the results depend, to some extent, on the specific method that has been used for analysis of data. We have analysed height and growth velocity data as well as the applied methodology from 13 studies on spontaneous growth in UTS. Most studies were based on calculations of annual means or medians of data collected in a longitudinal/cross-sectional manner; others used mathematical models. Growth velocities were calculated longitudinally in individual patients or were derived from height curves graphically or mathematically. Individual height data for a given age varied between 2.6 and 7.7 cm, when estimated annual means were applied, and between 0.4 and 5.8 cm when mathematical models were used. Reported data on growth velocity were almost identical in all studies except for the age of expected puberty, when some authors found a minor pubertal growth spurt. Standard deviations for growth velocity increased at the time of pubertal age and amounted to up to 70% of the respective growth velocity. When various UTS height standards were applied for evaluation of treatment effects, we found a difference of up to 100% of the SD score due to the different SD values of reference data. Results expressed as height SD score may be biased by relatively low mean heights of reference data at adolescent ages.
患有乌尔里希-特纳综合征(UTS)的患者越来越多地接受生长激素(GH)治疗,以提高生长速度和最终身高。为了评估这种治疗的效果,人们应用了不同的方法,而我们证明,结果在一定程度上取决于用于数据分析的具体方法。我们分析了13项关于UTS自然生长的研究中的身高和生长速度数据以及所应用的方法。大多数研究基于对以纵向/横断面方式收集的数据的年度均值或中位数的计算;其他研究则使用数学模型。生长速度是在个体患者中纵向计算的,或者是通过图形或数学方法从身高曲线中得出的。当应用估计的年度均值时,给定年龄的个体身高数据在2.6至7.7厘米之间变化,而当使用数学模型时,在0.4至5.8厘米之间变化。除了预期青春期年龄外,所有研究中报告的生长速度数据几乎相同,一些作者在该年龄发现了轻微的青春期生长突增。青春期时生长速度的标准差增加,高达各自生长速度的70%。当应用各种UTS身高标准来评估治疗效果时,由于参考数据的标准差不同,我们发现标准差得分的差异高达100%。以身高标准差得分表示的结果可能会因青少年时期参考数据的平均身高相对较低而产生偏差。