Servicio de Pediatría, Hospital General Universitario Gregorio Marañón, Instituto de Investigación Sanitaria Gregorio Marañón (IiSGM), Madrid, Spain.
Servicio de Pediatría, Hospital Universitario de Cruces, Barakaldo, Bizkaia, Spain.
An Pediatr (Engl Ed). 2023 Dec;99(6):385-392. doi: 10.1016/j.anpede.2023.11.007. Epub 2023 Nov 21.
Primary ovarian insufficiency (POI) carries significant morbidity, causing infertility, sexual disfunction, decreased bone density, cardiovascular risk, emotional distress and early mortality.
To know the incidence and current management of POI in childhood/adolescent solid tumour survivors.
We conducted a multicentre observational study. It included female patients aged 12-18 years with a diagnosis of solid tumour and meeting clinical or biochemical criteria for POI. The risk was estimated based on the criteria of the Pediatric Initiative Network of the Oncofertility Consortium.
We found an incidence of 1.5 (30 cases of POI): The median age at the time of the event was 14 years (standard deviation, 2.09). The solid tumours associated most frequently with POI were Ewing sarcoma and brain and germ cell tumours. Eighty-three percent of patients did not undergo fertility preservation. Sixty-three percent reported not having undergone menarche at the time of ovarian failure. Ninety-seven percent were at high risk of gonadal toxicity, yet 47% were not monitored before the diagnosis. The median time elapsed to the occurrence of the event was 43.5 months after diagnosis and 29.5 months after completing treatment. The Kaplan-Meier curves showed that approximately 30% of POI cases developed within 2 years of diagnosis and that women at Tanner stage 1 developed insufficiency later than women at Tanner stage 5.
There is room for improvement in the follow-up of women at risk of POI in Spain. The tools currently available facilitate risk assessment at the time of treatment planning and allow the implementation of monitoring, education, early diagnosis, fertility preservation, and replacement therapy as needed. All of this would achieve significant improvement in health outcomes.
原发性卵巢功能不全(POI)会导致显著的发病率,引起不孕、性功能障碍、骨密度降低、心血管风险、情绪困扰和早逝。
了解儿童/青少年实体瘤幸存者中 POI 的发生率和当前管理情况。
我们进行了一项多中心观察性研究。纳入年龄在 12-18 岁、患有实体瘤且符合 POI 临床或生化标准的女性患者。风险是根据儿科肿瘤生育力倡议网络的标准进行估计的。
我们发现 POI 的发生率为 1.5(30 例 POI):事件发生时的中位年龄为 14 岁(标准差为 2.09)。与 POI 相关的最常见实体瘤是尤文肉瘤和脑及生殖细胞肿瘤。83%的患者未进行生育力保存。63%的患者在卵巢衰竭时报告尚未出现初潮。97%的患者有发生性腺毒性的高风险,但 47%的患者在诊断前未接受监测。从诊断到事件发生的中位时间为诊断后 43.5 个月,治疗结束后 29.5 个月。Kaplan-Meier 曲线显示,大约 30%的 POI 病例在诊断后 2 年内发生,Tanner 1 期的女性比 Tanner 5 期的女性发生功能不全的时间晚。
西班牙对 POI 风险女性的随访有待改善。目前可用的工具有助于在治疗计划时进行风险评估,并允许根据需要进行监测、教育、早期诊断、生育力保存和替代治疗,所有这些都将显著改善健康结果。