Abstract：
Should we perform oocyte accumulation to preserve fertility in women with Turner syndrome (TS)?
The oocyte cryopreservation strategy is not well adapted for all TS women as their combination of high basal FSH with low basal AMH and low percentage of 46,XX cells in the karyotype significantly reduces the chances of freezing sufficient mature oocytes for fertility preservation.
An oocyte cryopreservation strategy requiring numerous stimulation cycles is needed to preserve fertility in TS women, to compensate for the low ovarian response, the possible oocyte genetic alterations, the reduced endometrial receptivity, and the increased rate of miscarriage, observed in this specific population. The validation of reliable predictive biomarkers of ovarian response to hormonal stimulation in TS patients is necessary to help practitioners and patients choose the best-personalized fertility preservation strategy.
A retrospective bicentric study was performed from 1 January 2011 to 1 January 2023. Clinical and biological data from all TS women who have received from ovarian stimulation for fertility preservation were collected. A systematic review of the current literature on oocyte retrieval outcomes after ovarian stimulation in TS women was also performed (PROSPERO registration number: CRD42022362352).
A total of 14 TS women who had undergone ovarian stimulation for fertility preservation were included, representing the largest cohort of TS patients published to date (n = 14 patients, 24 cycles). The systematic review of the literature identified 34 additional TS patients with 47 oocyte retrieval outcomes after ovarian stimulation in 14 publications (n = 48 patients, n = 71 cycles in total).
The number of cryopreserved mature oocytes on the first cycle for TS patients was low (4.0 ± 3.7). Oocyte accumulation was systematically proposed to increase fertility potential and was accepted by 50% (7/14) of patients (2.4 ± 0.5 cycles), leading to an improved total number of 10.9 ± 7.2 cryopreserved mature oocytes per patient. In the group who refused the oocyte accumulation strategy, only one patient exceeded the threshold of 10 mature cryopreserved oocytes. In contrast, 57.1% (4/7) and 42.9% (3/7) of patients who have underwent the oocyte accumulation strategy reached the threshold of 10 and 15 mature cryopreserved oocytes, respectively (OR = 8 (0.6; 107.0), P = 0.12; OR= 11 (0.5; 282.1), P = 0.13). By analyzing all the data published to date and combining it with our data (n = 48 patients, n = 71 cycles), low basal FSH and high AMH concentrations as well as a higher percentage of 46,XX cells in the karyotype were significantly associated with a higher number of cryopreserved oocytes after the first cycle. Moreover, the combination of low basal FSH concentration (<5.9 IU/l), high AMH concentration (>1.13 ng/ml), and the presence of 46,XX cells (>1%) was significantly predictive of obtaining at least six cryopreserved oocytes in the first cycle, representing objective criteria for identifying patients with real chances of preserving an adequate fertility potential by oocyte cryopreservation.
Our results should be analyzed with caution, as the optimal oocyte number needed for successful live birth in TS patients is still unknown due to the low number of reports their oocyte use in the literature to date.
TS patients should benefit from relevant clinical evaluation, genetic counseling and psychological support to make an informed choice regarding their fertility preservation technique, as numerous stimulation cycles would be necessary to preserve a high number of oocytes.
This research received no external funding. The authors declare no conflict of interest.
N/A.
The oocyte cryopreservation strategy is not well adapted for all TS women as their combination of high basal FSH with low basal AMH and low percentage of 46,XX cells in the karyotype significantly reduces the chances of freezing sufficient mature oocytes for fertility preservation.
An oocyte cryopreservation strategy requiring numerous stimulation cycles is needed to preserve fertility in TS women, to compensate for the low ovarian response, the possible oocyte genetic alterations, the reduced endometrial receptivity, and the increased rate of miscarriage, observed in this specific population. The validation of reliable predictive biomarkers of ovarian response to hormonal stimulation in TS patients is necessary to help practitioners and patients choose the best-personalized fertility preservation strategy.
A retrospective bicentric study was performed from 1 January 2011 to 1 January 2023. Clinical and biological data from all TS women who have received from ovarian stimulation for fertility preservation were collected. A systematic review of the current literature on oocyte retrieval outcomes after ovarian stimulation in TS women was also performed (PROSPERO registration number: CRD42022362352).
A total of 14 TS women who had undergone ovarian stimulation for fertility preservation were included, representing the largest cohort of TS patients published to date (n = 14 patients, 24 cycles). The systematic review of the literature identified 34 additional TS patients with 47 oocyte retrieval outcomes after ovarian stimulation in 14 publications (n = 48 patients, n = 71 cycles in total).
The number of cryopreserved mature oocytes on the first cycle for TS patients was low (4.0 ± 3.7). Oocyte accumulation was systematically proposed to increase fertility potential and was accepted by 50% (7/14) of patients (2.4 ± 0.5 cycles), leading to an improved total number of 10.9 ± 7.2 cryopreserved mature oocytes per patient. In the group who refused the oocyte accumulation strategy, only one patient exceeded the threshold of 10 mature cryopreserved oocytes. In contrast, 57.1% (4/7) and 42.9% (3/7) of patients who have underwent the oocyte accumulation strategy reached the threshold of 10 and 15 mature cryopreserved oocytes, respectively (OR = 8 (0.6; 107.0), P = 0.12; OR= 11 (0.5; 282.1), P = 0.13). By analyzing all the data published to date and combining it with our data (n = 48 patients, n = 71 cycles), low basal FSH and high AMH concentrations as well as a higher percentage of 46,XX cells in the karyotype were significantly associated with a higher number of cryopreserved oocytes after the first cycle. Moreover, the combination of low basal FSH concentration (<5.9 IU/l), high AMH concentration (>1.13 ng/ml), and the presence of 46,XX cells (>1%) was significantly predictive of obtaining at least six cryopreserved oocytes in the first cycle, representing objective criteria for identifying patients with real chances of preserving an adequate fertility potential by oocyte cryopreservation.
Our results should be analyzed with caution, as the optimal oocyte number needed for successful live birth in TS patients is still unknown due to the low number of reports their oocyte use in the literature to date.
TS patients should benefit from relevant clinical evaluation, genetic counseling and psychological support to make an informed choice regarding their fertility preservation technique, as numerous stimulation cycles would be necessary to preserve a high number of oocytes.
This research received no external funding. The authors declare no conflict of interest.
N/A.
摘要：
目的：我们是否应该在特纳综合征（TS）女性中进行卵母细胞积累以保持生育能力？
结论：卵母细胞冷冻保存策略并不能很好地适用于所有TS女性，因为它们的基础FSH高，基础AMH低，核型中46,XX细胞的百分比低，可以显着降低冷冻足够成熟卵母细胞以保持生育能力的机会。
背景：需要一种需要多次刺激周期的卵母细胞冷冻保存策略来保持TS女性的生育能力，为了补偿低卵巢反应，可能的卵母细胞遗传改变，子宫内膜容受性降低，流产率的增加，在这个特定人群中观察到。TS患者卵巢对激素刺激反应的可靠预测性生物标志物的验证对于帮助从业者和患者选择最佳的个性化生育力保存策略是必要的。
方法：2011年1月1日至2023年1月1日进行了一项回顾性双中心研究。收集了所有接受卵巢刺激以保持生育力的TS妇女的临床和生物学数据。本研究还对目前有关TS女性卵巢刺激后卵母细胞取出结果的文献进行了系统评价（PROSPERO注册号：CRD42022362352）。
方法：共包括14名接受卵巢刺激以保留生育力的TS妇女，代表迄今为止发表的最大的TS患者队列(n=14名患者，24个周期)。文献的系统评价在14篇出版物中确定了另外34例TS患者在卵巢刺激后有47个卵母细胞取出结果（n=48例，n=总共71个循环)。
结果：TS患者第一周期冷冻保存的成熟卵母细胞数量较少（4.0±3.7）。系统地提出了卵母细胞积累以增加生育潜力，并被50％（7/14）的患者接受（2.4±0.5个周期），导致每个患者冷冻保存的成熟卵母细胞总数增加10.9±7.2。在拒绝卵母细胞积累策略的组中，只有一名患者超过10个成熟冷冻保存卵母细胞的阈值.相比之下,57.1％（4/7）和42.9％（3/7）的患者进行了卵母细胞积累策略，达到了10和15个成熟冷冻保存卵母细胞的阈值，分别(OR=8(0.6;107.0)，P=0.12；OR=11（0.5；282.1），P=0.13）。通过分析迄今为止发表的所有数据，并将其与我们的数据(n=48名患者，n=71个循环)，低基础FSH和高AMH浓度以及核型中46,XX细胞的较高百分比与第一个周期后冷冻保存的卵母细胞数量显着相关。此外，低基础FSH浓度（<5.9IU/l）的组合，高AMH浓度（>1.13ng/ml），46，XX细胞（>1％）的存在显着预测在第一个周期中获得至少6个冷冻保存的卵母细胞，代表通过卵母细胞冷冻保存确定有真正机会保留足够生育潜力的患者的客观标准。
结论：我们的结果应谨慎分析，由于迄今为止文献中使用卵母细胞的报道数量很少，因此TS患者成功活产所需的最佳卵母细胞数量仍然未知。
结论：TS患者应该从相关的临床评估中获益，遗传咨询和心理支持，以做出明智的选择，关于他们的生育保存技术，因为许多刺激周期对于保存大量卵母细胞是必要的。
背景：这项研究没有获得外部资助。作者声明没有利益冲突。
背景：不适用。
结论：卵母细胞冷冻保存策略并不能很好地适用于所有TS女性，因为它们的基础FSH高，基础AMH低，核型中46,XX细胞的百分比低，可以显着降低冷冻足够成熟卵母细胞以保持生育能力的机会。
背景：需要一种需要多次刺激周期的卵母细胞冷冻保存策略来保持TS女性的生育能力，为了补偿低卵巢反应，可能的卵母细胞遗传改变，子宫内膜容受性降低，流产率的增加，在这个特定人群中观察到。TS患者卵巢对激素刺激反应的可靠预测性生物标志物的验证对于帮助从业者和患者选择最佳的个性化生育力保存策略是必要的。
方法：2011年1月1日至2023年1月1日进行了一项回顾性双中心研究。收集了所有接受卵巢刺激以保持生育力的TS妇女的临床和生物学数据。本研究还对目前有关TS女性卵巢刺激后卵母细胞取出结果的文献进行了系统评价（PROSPERO注册号：CRD42022362352）。
方法：共包括14名接受卵巢刺激以保留生育力的TS妇女，代表迄今为止发表的最大的TS患者队列(n=14名患者，24个周期)。文献的系统评价在14篇出版物中确定了另外34例TS患者在卵巢刺激后有47个卵母细胞取出结果（n=48例，n=总共71个循环)。
结果：TS患者第一周期冷冻保存的成熟卵母细胞数量较少（4.0±3.7）。系统地提出了卵母细胞积累以增加生育潜力，并被50％（7/14）的患者接受（2.4±0.5个周期），导致每个患者冷冻保存的成熟卵母细胞总数增加10.9±7.2。在拒绝卵母细胞积累策略的组中，只有一名患者超过10个成熟冷冻保存卵母细胞的阈值.相比之下,57.1％（4/7）和42.9％（3/7）的患者进行了卵母细胞积累策略，达到了10和15个成熟冷冻保存卵母细胞的阈值，分别(OR=8(0.6;107.0)，P=0.12；OR=11（0.5；282.1），P=0.13）。通过分析迄今为止发表的所有数据，并将其与我们的数据(n=48名患者，n=71个循环)，低基础FSH和高AMH浓度以及核型中46,XX细胞的较高百分比与第一个周期后冷冻保存的卵母细胞数量显着相关。此外，低基础FSH浓度（<5.9IU/l）的组合，高AMH浓度（>1.13ng/ml），46，XX细胞（>1％）的存在显着预测在第一个周期中获得至少6个冷冻保存的卵母细胞，代表通过卵母细胞冷冻保存确定有真正机会保留足够生育潜力的患者的客观标准。
结论：我们的结果应谨慎分析，由于迄今为止文献中使用卵母细胞的报道数量很少，因此TS患者成功活产所需的最佳卵母细胞数量仍然未知。
结论：TS患者应该从相关的临床评估中获益，遗传咨询和心理支持，以做出明智的选择，关于他们的生育保存技术，因为许多刺激周期对于保存大量卵母细胞是必要的。
背景：这项研究没有获得外部资助。作者声明没有利益冲突。
背景：不适用。
