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抗雄激素、芳香化酶抑制剂和促性腺激素释放激素类似物对家族性男性性早熟成年身高的影响。

Effect of Antiandrogen, Aromatase Inhibitor, and Gonadotropin-releasing Hormone Analog on Adult Height in Familial Male Precocious Puberty.

作者信息

Leschek Ellen Werber, Flor Armando C, Bryant Joy C, Jones Janet V, Barnes Kevin M, Cutler Gordon B

机构信息

National Institute of Diabetes and Digestive and Kidney Diseases, National Institutes of Health, Bethesda, MD.

MedImmune, Gaithersburg, MD.

出版信息

J Pediatr. 2017 Nov;190:229-235. doi: 10.1016/j.jpeds.2017.07.047.

Abstract

OBJECTIVE

Antiandrogen, aromatase inhibitor, and gonadotropin-releasing hormone analog (GnRHa) treatment normalizes growth rate and bone maturation and increases predicted adult height (AH) in boys with familial male-limited precocious puberty (FMPP). To evaluate the effect of long-term antiandrogen, aromatase inhibitor, and GnRHa on AH, boys with FMPP who were treated were followed to AH.

STUDY DESIGN

Twenty-eight boys with FMPP, referred to the National Institutes of Health, were started on antiandrogen and aromatase inhibitor at 4.9 ± 1.5 years of age; GnRHa was added at 6.9 ± 1.5 years of age. Treatment was discontinued at 12.2 ± 0.5 years of age (bone age, 14.4 ± 1.3). AH was assessed at 16.4 ± 1.3 years of age (bone age, 18.5 ± 0.6).

RESULTS

AH (mean ± SD) for all treated subjects was 173.6 ± 6.8 cm (-0.4 ± 1.0 SD relative to adult US males). For 25 subjects with pretreatment predicted AH, AH significantly exceeded predicted AH at treatment onset (173.8 ± 6.9 vs 164.9 ± 10.7 cm; P < .001), but fell short of predicted AH at treatment discontinuation (177.3 ± 9.0 cm; P < .001). For 11 subjects with maternal or sporadic inheritance, the mean AH was 3.1 cm (0.4 SD score) below sex-adjusted midparental height (175.4 ± 5.8 vs 178.5 ± 3.1 cm [midparental height]; P = .10). For 16 subjects with affected and untreated fathers, AH was significantly greater than fathers' AH (172.8 ± 7.4 vs 168.8 ± 7.2 cm; P < .05).

CONCLUSIONS

Long-term treatment with antiandrogen, aromatase inhibitor, and GnRHa in boys with FMPP results in AH modestly below sex-adjusted midparental height and within the range for adult males in the general population.

摘要

目的

抗雄激素、芳香化酶抑制剂和促性腺激素释放激素类似物(GnRHa)治疗可使家族性男性性早熟(FMPP)男孩的生长速率和骨骼成熟正常化,并增加预测成年身高(AH)。为评估长期使用抗雄激素、芳香化酶抑制剂和GnRHa对AH的影响,对接受治疗的FMPP男孩进行随访直至成年身高。

研究设计

28名转诊至美国国立卫生研究院的FMPP男孩在4.9±1.5岁时开始使用抗雄激素和芳香化酶抑制剂治疗;6.9±1.5岁时加用GnRHa。治疗在12.2±0.5岁(骨龄14.4±1.3)时停止。在16.4±1.3岁(骨龄18.5±0.6)时评估AH。

结果

所有接受治疗的受试者的AH(均值±标准差)为173.6±6.8cm(相对于美国成年男性,标准差为-0.4±1.0)。对于25名有治疗前预测AH的受试者,AH在治疗开始时显著超过预测AH(173.8±6.9 vs 164.9±10.7cm;P<.001),但在治疗停止时低于预测AH(177.3±9.0cm;P<.001)。对于11名有母系或散发性遗传的受试者,平均AH比按性别调整的父母平均身高低3.1cm(标准差分数为0.4)(175.4±5.8 vs 178.5±3.1cm[父母平均身高];P=.10)。对于16名父亲患病且未接受治疗的受试者,AH显著高于父亲的AH(172.8±7.4 vs 168.8±7.2cm;P<.05)。

结论

FMPP男孩长期使用抗雄激素、芳香化酶抑制剂和GnRHa治疗后,AH略低于按性别调整的父母平均身高,处于一般人群成年男性的范围内。

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