Wang Xi, Yu Qi
Department of Obstetrics and Gynaecology, Peking Union Medical College Hospital, Peking Union Medical College and Chinese Academy of Medical Sciences, Beijing, China.
Endocr Connect. 2018 Dec 1;7(12):1424-1431. doi: 10.1530/EC-18-0344.
Objective To evaluate the safety and efficacy of letrozole in girls with progressive precocious puberty (PP) associated with McCune-Albright syndrome (MAS). Design Monocentric retrospective cross-sectional and longitudinal study of consecutive patients. Patients Ten MAS patients treated at Peking Union Medical College Hospital between September 1999 and December 2017 were retrospectively reviewed; those with complications due to PP were followed. Results The mean age at letrozole initiation was 4.5 ± 2.6 years, while the mean duration of treatment was 3.3 ± 2.4 years. Letrozole was highly effective at decreasing the rate of skeletal maturation, with a significant decrease in the bone age-to-chronological age (BA/CA) ratio from 1.9 ± 1.1 pre-treatment to 1.5 ± 1.2 on letrozole treatment (P = 0.016). Moreover, growth velocity Z-scores declined from 0.41 ± 0.5 to -0.2 ± 0.31 with treatment (P < 0.001). Predicted adult height Z-scores increased significantly from -2.03 ± 2.33 at baseline to 1.13 ± 0.84 following treatment initiation (P = 0.029). Moreover, vaginal bleeding declined significantly on letrozole. Conclusions Our findings suggest that letrozole may be an effective therapy in some girls with MAS, as treatment results in improved BA/CA ratio, growth velocity and predicted adult height. Possible adverse effects include nettle rash.
目的 评估来曲唑治疗与McCune - Albright综合征(MAS)相关的进行性性早熟(PP)女童的安全性和有效性。设计 对连续患者进行单中心回顾性横断面和纵向研究。患者 回顾性分析1999年9月至2017年12月在北京协和医院接受治疗的10例MAS患者;对因PP出现并发症的患者进行随访。结果 开始使用来曲唑时的平均年龄为4.5±2.6岁,平均治疗时长为3.3±2.4年。来曲唑在降低骨骼成熟速率方面非常有效,骨龄与实际年龄(BA/CA)比从治疗前的1.9±1.1显著降至来曲唑治疗时的1.5±1.2(P = 0.016)。此外,治疗后生长速度Z值从0.41±0.5降至 - 0.2±0.31(P < 0.001)。预测的成人身高Z值从基线时的 - 2.03±2.33显著增至开始治疗后的1.13±0.84(P = 0.029)。而且,来曲唑治疗后阴道出血显著减少。结论 我们的研究结果表明,来曲唑可能是治疗部分MAS女童的有效疗法,因为治疗可改善BA/CA比、生长速度和预测的成人身高。可能的不良反应包括荨疹。