Department of Pediatrics and Developmental Biology, Tokyo Medical and Dental University, Tokyo 113-8510, Japan.
Endocr J. 2012;59(11):1001-6. doi: 10.1507/endocrj.ej12-0036. Epub 2012 Jul 10.
An initial high-dose treatment of glucocorticoid has been proposed to prevent chronic androgen excess, improving the final height prognosis of 21-hydroxylase deficiency (21-OHD) patients. In Japan, it is recommended to use an extremely high-dose of hydrocortisone (HDC) (100-200 mg/m(2)/day) for initial treatment by the Japanese Society for Pediatric Endocrinology. However, a precise evaluation of the treatment has not been carried out. In this study, we retrospectively analysed the effects of initial high-dose HDC therapy on the linear growth of classical 21-OHD patients discovered by newborn screening. Thirty patients (14 females) were eligible for this study, all of whom were initiated with high dose HDC therapy. The height standard deviation score (Ht-SDS) was 0.76 ± 0.65 at birth, and decreased to -1SD or less until the age of 12 months, subsequently catching up by 3 years of age (-0.56 ± 0.76). The growth pattern and the height at the age of two years were very similar to those previously observed in patients without initial high dose HDC therapy. We did not find any significant difference in growth retrospectively between the high- or low-dose HDC group (initial treatments of ≥150 mg/m(2)/day and 100 mg/m(2)/day, respectively). Bone ages did not exceed chronological ages at the ages of three and six years. Our data suggest that an initial high-dose HDC treatment does not profoundly affect linear growth during first three years of life and that the treatment could be a valuable option for 21-OHD patients without having an obvious adverse effect on linear growth.
初始高剂量糖皮质激素治疗被提议用于预防慢性雄激素过多,改善 21-羟化酶缺乏症(21-OHD)患者的最终身高预测。在日本,日本儿科内分泌学会建议使用极高剂量的氢化可的松(HDC)(100-200mg/m²/天)进行初始治疗。然而,尚未对该治疗进行精确评估。在这项研究中,我们回顾性分析了初始高剂量 HDC 治疗对通过新生儿筛查发现的经典 21-OHD 患者线性生长的影响。符合条件的 30 名患者(14 名女性)均接受了高剂量 HDC 治疗。出生时身高标准差评分(Ht-SDS)为 0.76±0.65,12 个月龄时降至-1SD 或更低,随后在 3 岁时追上(-0.56±0.76)。生长模式和 2 岁时的身高与之前观察到的无初始高剂量 HDC 治疗的患者非常相似。我们没有发现回顾性生长在高剂量或低剂量 HDC 组之间有任何显著差异(初始治疗分别为≥150mg/m²/天和 100mg/m²/天)。骨龄在 3 岁和 6 岁时均未超过年龄。我们的数据表明,初始高剂量 HDC 治疗不会在生命的前三年显著影响线性生长,并且该治疗可能是 21-OHD 患者的一种有价值的选择,对线性生长没有明显的不良影响。