Puberty progression in girls with Turner syndrome after ovarian tissue cryopreservation.

IF 6.6 1区医学 Q1 OBSTETRICS & GYNECOLOGY

Fertility and sterility Pub Date : 2024-10-19 DOI:10.1016/j.fertnstert.2024.10.025

Sanne van der Coelen, Sapthami Nadesapillai, Ronald Peek, Didi Braat, Gianni Bocca, Martijn Finken, Sabine Hannema, Sandra de Kort, Theo Sas, Saartje Straetemans, Vera van Tellingen, Annemarie Verrijn Stuart, Kathrin Fleischer, Janielle van der Velden

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引用次数: 0

Abstract

Objective: To study the impact of unilateral ovariectomy for ovarian tissue cryopreservation (OTC) on the function of the remaining ovary in girls with Turner syndrome.

Design: A prospective cohort study as follow up of OTC in a research setting (The TurnerFertility trial, NCT03381300).

Setting: University hospital in Nijmegen, the Netherlands.

Subjects: A total of 28 girls with Turner syndrome with follicles in their cryopreserved ovarian cortex tissue, aged 5-19 years. Of the 28 girls, 21 girls had a 45,X/46,XX mosaic karyotype, five had structural aberrations of the X-chromosome, one girl had a 45,X monosomy, and one girl had a 45,X/47,XXX karyotype.

Interventions: Girls were monitored annually after OTC for pubertal development and levels of anti-Müllerian hormone (AMH), Follicle Stimulating Hormone, Luteinizing Hormone, Estradiol, and Inhibin B.

Main outcome measures: Thelarche, menarche and onset of premature ovarian insufficiency.

Results: The girls were monitored for a median duration of 3.4 years (maximum 6.6 years). The pubertal development of five prepubertal girls is still unknown, all were under the age of 10 and had low gonadotropins and estradiol levels at the end of the follow-up. Seven of the eight girls around pubertal age (10-12 years) experienced spontaneous thelarche, while one received medication to induce puberty. Eleven of the fourteen girls between the age of 14-17 years experienced spontaneous menarche, three other girls with thelarche still had ongoing puberty at the end of follow-up with normal gonadotropins and AMH levels above the detection limit. Around six to twelve months after OTC, a decline in AMH concentration was observed in 57% of girls (16/28 girls), followed by an increase in AMH concentration in the following years. Six of the total 28 girls started hormone replacement therapy due to symptoms of premature ovarian insufficiency, and all had AMH levels below 0.50 μg/L before OTC.

Conclusion: Pubertal development progressed after unilateral ovariectomy for OTC in most girls with Turner syndrome. Hormone replacement therapy was required within a few years for girls with unfavorable parameters before OTC, such as AMH below 0.50 μg/L. Decisions regarding OTC should be personalized, considering the girl's preferences and specific characteristics.

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特纳综合征女孩卵巢组织冷冻后的青春期进展。

目的：研究单侧卵巢切除术（OTC）对特纳综合征女孩剩余卵巢功能的影响：研究单侧卵巢切除术进行卵巢组织冷冻保存（OTC）对特纳综合征女孩剩余卵巢功能的影响：设计：一项前瞻性队列研究，作为在研究环境中对 OTC 进行的随访（TurnerFertility 试验，NCT03381300）：地点：荷兰奈梅亨大学医院：共有 28 名患有特纳综合征的女孩，其冷冻保存的卵巢皮质组织中有卵泡，年龄为 5-19 岁。28名女孩中，21名女孩的核型为45,X/46,XX镶嵌型，5名女孩的X染色体结构畸变，1名女孩的核型为45,X单体型，1名女孩的核型为45,X/47,XXX型：干预措施：在 OTC 后，每年监测女孩的青春期发育情况以及抗缪勒氏管激素（AMH）、卵泡刺激素、促黄体生成素、雌二醇和抑制素 B 的水平：主要结果指标：初潮、月经初潮和卵巢早衰：对这些女孩的监测时间中位数为 3.4 年（最长 6.6 年）。五名青春期前女孩的青春期发育情况尚不清楚，她们都在 10 岁以下，在随访结束时促性腺激素和雌二醇水平较低。八名青春期前后（10-12 岁）的女孩中，七名自发月经初潮，一名接受药物诱导青春期发育。在 14-17 岁的 14 名女孩中，有 11 人自然初潮，另外 3 名初潮女孩在随访结束时仍处于青春期，促性腺激素正常，AMH 水平高于检测限。在 OTC 后的 6 至 12 个月左右，57% 的女孩（16/28 名女孩）的 AMH 浓度下降，随后几年 AMH 浓度上升。在总共 28 名女孩中，有 6 名女孩因出现卵巢早衰症状而开始接受激素替代治疗，她们在接受 OTC 治疗前的 AMH 水平均低于 0.50 μg/L：结论：大多数特纳综合征女孩在因 OTC 而进行单侧卵巢切除术后，青春期发育会有所进展。结论：大多数特纳综合征女孩在接受单侧卵巢切除术后，青春期发育会有所进展。对于那些在接受 OTC 之前各项指标不佳（如 AMH 低于 0.50 μg/L）的女孩，需要在几年内接受激素替代治疗。考虑到女孩的偏好和具体特征，有关 OTC 的决定应个性化。

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来源期刊

Fertility and sterility 医学-妇产科学

CiteScore

11.30

自引率

6.00%

发文量

1446

审稿时长

31 days

期刊介绍： Fertility and Sterility® is an international journal for obstetricians, gynecologists, reproductive endocrinologists, urologists, basic scientists and others who treat and investigate problems of infertility and human reproductive disorders. The journal publishes juried original scientific articles in clinical and laboratory research relevant to reproductive endocrinology, urology, andrology, physiology, immunology, genetics, contraception, and menopause. Fertility and Sterility® encourages and supports meaningful basic and clinical research, and facilitates and promotes excellence in professional education, in the field of reproductive medicine.