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先天性巨结肠症女性的生育力和性功能受损:一项国际多中心横断面研究的结果

Impaired Fertility and Sexual Function in Women With Hirschsprung Disease: Results From an International Multi-Centre Cross-Sectional Study.

作者信息

Davidson Joseph R, Mutanen Annika, Granström Anna Löf, Hoel Anders Telle, Loukogeorgakis Stavros P, De Coppi Paolo, Bjørnland Kristin, Wester Tomas, Eaton Simon, Pakarinen Mikko P, Curry Joe

机构信息

Great Ormond Street Hospital for Children, London, UK.

UCL Great Ormond Street Institute of Child Health, London, UK.

出版信息

BJOG. 2025 Oct;132(11):1673-1680. doi: 10.1111/1471-0528.18294. Epub 2025 Jul 10.

DOI:10.1111/1471-0528.18294
PMID:40637045
Abstract

OBJECTIVE

Hirschsprung is a congenital disorder affecting the gastrointestinal tract. However, pelvic colorectal surgery in infancy has been hypothesised to impact gynaecological outcomes in later life. Describe sexual function and fertility outcomes in women with Hirschsprung disease compared to population controls. Assess factors associated with poor outcomes (sexual dysfunction and subfertility).

DESIGN

International multicentre cross-sectional cohort study with comparison to controls from the general population.

SETTING

Status post-discharge from paediatric services.

POPULATION

Female patients aged > 20 years.

METHODS

Validated questionnaire-based survey with linkage to patient medical records. Comparison with controls using univariate analyses.

MAIN OUTCOME MEASURES

Sexual dysfunction (Female Sexual Function Index; FSFI ≤ 26), Subfertility at 1 and 2 years.

RESULTS

Sexual dysfunction as per the FSFI was more common in patients and associated with poor functional outcomes; sexual abstinence seemed to associate even more so with poor bowel outcomes. Subfertility was higher in patients compared to controls (1 year: 21/45 (47%) vs. 38/178 (21%), p = 0.0008; 2 years: 12/45 (27%) vs. 17/178 (10%), p = 0.004). There was an increased proportion of patients who had accessed fertility services (20/45 (44%) vs. 43/178 (24%); p = 0.009), the proportion of successful pregnancies in patients attempting to conceive with IVF (11/17 (65%) vs. 27/43 (63%); p = 1.0) was similar.

CONCLUSIONS

These novel data suggest that women with Hirschsprung disease who have undergone reconstructive surgery may be at risk for adverse sexual function and fertility outcomes.

摘要

目的

先天性巨结肠是一种影响胃肠道的先天性疾病。然而,有假说认为婴儿期的盆腔结直肠手术会对其成年后的妇科结局产生影响。描述先天性巨结肠病女性与一般人群对照者的性功能和生育结局。评估与不良结局(性功能障碍和生育力低下)相关的因素。

设计

国际多中心横断面队列研究,并与一般人群对照者进行比较。

背景

儿科服务出院后状态。

研究对象

年龄大于20岁的女性患者。

方法

基于问卷的有效调查,并与患者病历相联系。采用单因素分析与对照者进行比较。

主要观察指标

性功能障碍(女性性功能指数;FSFI≤26)、1年和2年时的生育力低下。

结果

根据FSFI,性功能障碍在患者中更常见,且与不良功能结局相关;性禁欲似乎与不良肠道结局的关联更强。与对照者相比,患者的生育力低下情况更严重(1年时:21/45(47%)对38/178(21%),p = 0.0008;2年时:12/45(27%)对17/178(10%),p = 0.004)。使用生育服务的患者比例增加(20/45(44%)对43/178(24%);p = 0.009),尝试通过体外受精受孕的患者中成功妊娠的比例(11/17(65%)对27/43(63%);p = 1.0)相似。

结论

这些新数据表明,接受过重建手术的先天性巨结肠病女性可能面临性功能和生育结局不良的风险。

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本文引用的文献

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Transitional Care in Patients With Hirschsprung Disease: Those Left Behind.先天性巨结肠症患者的过渡性护理:被忽视的群体。
Dis Colon Rectum. 2024 Jul 1;67(7):977-984. doi: 10.1097/DCR.0000000000003208. Epub 2024 Apr 23.
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Fertility in patients with Hirschsprung's disease: population-based cohort study.先天性巨结肠症患者的生育能力:基于人群的队列研究。
BJS Open. 2023 May 5;7(3). doi: 10.1093/bjsopen/zrad043.
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Comparative cohort study of Duhamel and endorectal pull-through for Hirschsprung's disease.Duhamel 与经肛门内拖出术治疗先天性巨结肠的对比队列研究。
BJS Open. 2022 Jan 6;6(1). doi: 10.1093/bjsopen/zrab143.
4
Sexual function, quality of life, and fertility in women who had surgery for neonatal Hirschsprung's disease.接受新生儿先天性巨结肠手术的女性的性功能、生活质量和生育能力。
Br J Surg. 2021 Mar 12;108(2):e79-e80. doi: 10.1093/bjs/znaa108.
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Long-term surgical and patient-reported outcomes of Hirschsprung Disease.先天性巨结肠症的长期手术和患者报告结果。
J Pediatr Surg. 2021 Sep;56(9):1502-1511. doi: 10.1016/j.jpedsurg.2021.01.043. Epub 2021 Feb 13.
6
Evaluation of Bowel Function, Urinary Tract Function, and Quality of Life after Transanal Endorectal Pull-Through Surgery for Hirschsprung's Disease.经肛门内括约肌切除拖出术治疗先天性巨结肠后肠功能、尿路功能及生活质量的评估。
Eur J Pediatr Surg. 2021 Feb;31(1):40-48. doi: 10.1055/s-0040-1715612. Epub 2020 Sep 2.
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Reaching adulthood with Hirschsprung's disease: Patient experiences and recommendations for transitional care.成年期先天性巨结肠症患者:患者经历及过渡护理建议。
J Pediatr Surg. 2021 Feb;56(2):257-262. doi: 10.1016/j.jpedsurg.2020.05.015. Epub 2020 May 16.
8
"Too much guts and not enough brains": (epi)genetic mechanisms and future therapies of Hirschsprung disease - a review.“太多的勇气,而不是足够的头脑”:(表观遗传学)遗传机制与先天性巨结肠病的未来治疗方法——综述。
Clin Epigenetics. 2019 Sep 13;11(1):135. doi: 10.1186/s13148-019-0718-x.
9
Does laparoscopic ileal pouch-anal anastomosis reduce infertility compared with open approach?腹腔镜回肠贮袋肛管吻合术与开放手术相比,是否能降低不孕率?
Surgery. 2019 Oct;166(4):670-677. doi: 10.1016/j.surg.2019.04.045. Epub 2019 Aug 14.
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Overlooked Long-Term Complications of Colorectal Surgery.结直肠手术被忽视的长期并发症
Clin Colon Rectal Surg. 2019 May;32(3):204-211. doi: 10.1055/s-0038-1677027. Epub 2019 Apr 2.