• 文献检索
  • 文档翻译
  • 深度研究
  • 学术资讯
  • Suppr Zotero 插件Zotero 插件
  • 邀请有礼
  • 套餐&价格
  • 历史记录
应用&插件
Suppr Zotero 插件Zotero 插件浏览器插件Mac 客户端Windows 客户端微信小程序
定价
高级版会员购买积分包购买API积分包
服务
文献检索文档翻译深度研究API 文档MCP 服务
关于我们
关于 Suppr公司介绍联系我们用户协议隐私条款
关注我们

Suppr 超能文献

核心技术专利:CN118964589B侵权必究
粤ICP备2023148730 号-1Suppr @ 2026

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验

在1型戈谢病女性患者中,于艾加莫德临床试验期间意外怀孕的妊娠结局。

Pregnancy outcome in women with Gaucher disease type 1 who had unplanned pregnancies during eliglustat clinical trials.

作者信息

Lukina Elena, Balwani Manisha, Belmatoug Nadia, Watman Nora, Hughes Derralynn, Gaemers Sebastiaan J M, Foster Meredith C, Lewis Grace, Peterschmitt M Judith

机构信息

National Research Center for Hematology Moscow Russia.

Icahn School of Medicine at Mount Sinai New York New York USA.

出版信息

JIMD Rep. 2020 Oct 18;57(1):76-84. doi: 10.1002/jmd2.12172. eCollection 2021 Jan.

DOI:10.1002/jmd2.12172
PMID:33473343
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC7802626/
Abstract

Gaucher disease type 1 (GD1) is an inherited lysosomal storage disorder caused by deficient enzymatic activity of acid β-glucosidase, resulting in accumulation of its substrate glucosylceramide, leading to debilitating visceral, hematologic, and skeletal manifestations. Women with GD1 are at increased risk for complications during pregnancy, delivery, and postpartum. Treatment with enzyme replacement therapy is generally recommended before and during pregnancy to reduce risks. Eliglustat, an oral substrate-reduction therapy, is a first-line treatment for adults with GD1 adults who have extensive, intermediate, or poor CYP2D6-metabolizer phenotypes (>90% of patients). We report on pregnancy outcomes among women in eliglustat trials who had unplanned pregnancies and female partners of men in the trials. In four phase 2 and 3 eliglustat trials of 393 adults with GD1, women of childbearing potential were required to use contraception, have monthly pregnancy tests, and discontinue eliglustat promptly if pregnant. In phase 2 and 3 trials, 18 women had 19 pregnancies, resulting in 14 healthy infants from 13 pregnancies (one set of twins), three elective terminations, one ectopic pregnancy, one spontaneous abortion, and one in utero death. Median estimated eliglustat exposure duration during pregnancy was 38 days. In phase 1 trials (non-GD1 subjects), one woman had a spontaneous abortion. Partners of 16 eliglustat-treated men with GD1 had 18 pregnancies, all resulting in healthy infants. Eliglustat is not approved during pregnancy due to limited data. Guidelines for clinicians and patients with GD that address use of eliglustat in women of childbearing potential are needed.

摘要

1型戈谢病(GD1)是一种遗传性溶酶体贮积症,由酸性β-葡萄糖苷酶的酶活性缺乏引起,导致其底物葡萄糖神经酰胺蓄积,进而引发使人衰弱的内脏、血液学和骨骼表现。患有GD1的女性在妊娠、分娩和产后发生并发症的风险增加。一般建议在妊娠前和妊娠期间采用酶替代疗法进行治疗,以降低风险。艾加莫德,一种口服底物减少疗法,是具有广泛、中等或不良CYP2D6代谢表型的成年GD1患者(超过90%的患者)的一线治疗方法。我们报告了参与艾加莫德试验的意外怀孕女性以及试验中男性的女性伴侣的妊娠结局。在针对393名成年GD1患者的四项2期和3期艾加莫德试验中,有生育潜力的女性被要求采取避孕措施,每月进行妊娠试验,并且如果怀孕需立即停用艾加莫德。在2期和3期试验中,18名女性有19次妊娠,13次妊娠产下14名健康婴儿(其中一组双胞胎),3次选择性终止妊娠,1次异位妊娠,1次自然流产,1次子宫内死亡。妊娠期间估计的艾加莫德暴露持续时间中位数为38天。在1期试验(非GD1受试者)中,1名女性发生自然流产。16名接受艾加莫德治疗的患有GD1的男性的伴侣有18次妊娠,均产下健康婴儿。由于数据有限,艾加莫德在妊娠期间未获批准。需要针对临床医生和患有GD的患者制定关于有生育潜力女性使用艾加莫德的指南。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f550/7802626/0174a6ecd6f6/JMD2-57-76-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f550/7802626/0174a6ecd6f6/JMD2-57-76-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f550/7802626/0174a6ecd6f6/JMD2-57-76-g001.jpg

相似文献

1
Pregnancy outcome in women with Gaucher disease type 1 who had unplanned pregnancies during eliglustat clinical trials.在1型戈谢病女性患者中,于艾加莫德临床试验期间意外怀孕的妊娠结局。
JIMD Rep. 2020 Oct 18;57(1):76-84. doi: 10.1002/jmd2.12172. eCollection 2021 Jan.
2
Eliglustat: A Review in Gaucher Disease Type 1.依利格鲁司他:戈谢病 1 型治疗药物。
Drugs. 2015 Sep;75(14):1669-78. doi: 10.1007/s40265-015-0468-9.
3
Patient reported outcomes of patients with Gaucher disease type 1 treated with eliglustat in real-world settings: The ELIPRO study.在真实世界中接受依利格鲁司他治疗的1型戈谢病患者的患者报告结局:ELIPRO研究
Mol Genet Metab. 2023 Nov;140(3):107667. doi: 10.1016/j.ymgme.2023.107667. Epub 2023 Jul 26.
4
Eliglustat Therapy and Genotype依利格鲁司他疗法与基因型
5
Recommendations for oral treatment for adult patients with type 1 Gaucher disease.1型戈谢病成年患者的口服治疗建议。
Rev Clin Esp (Barc). 2022 Nov;222(9):529-542. doi: 10.1016/j.rceng.2022.02.008. Epub 2022 Jun 5.
6
Eliglustat tartrate for the treatment of adults with type 1 Gaucher disease.用于治疗成年1型戈谢病患者的酒石酸 eliglustat
Drug Des Devel Ther. 2015 Aug 18;9:4639-47. doi: 10.2147/DDDT.S77760. eCollection 2015.
7
Management and monitoring recommendations for the use of eliglustat in adults with type 1 Gaucher disease in Europe.欧洲1型戈谢病成年患者使用依利格鲁司他的管理和监测建议
Eur J Intern Med. 2017 Jan;37:25-32. doi: 10.1016/j.ejim.2016.07.011. Epub 2016 Aug 10.
8
Stability is maintained in adults with Gaucher disease type 1 switched from velaglucerase alfa to eliglustat or imiglucerase: A sub-analysis of the eliglustat ENCORE trial.从维拉苷酶α转换为依格列斯他或伊米苷酶的1型戈谢病成人患者的稳定性:依格列斯他ENCORE试验的亚分析
Mol Genet Metab Rep. 2016 Sep 30;9:25-28. doi: 10.1016/j.ymgmr.2016.08.009. eCollection 2016 Dec.
9
Once- versus twice-daily dosing of eliglustat in adults with Gaucher disease type 1: The Phase 3, randomized, double-blind EDGE trial.依利格鲁司他治疗 1 型戈谢病成人患者的每日一次与每日两次给药:III 期、随机、双盲 EDGE 试验。
Mol Genet Metab. 2018 Mar;123(3):347-356. doi: 10.1016/j.ymgme.2017.12.001. Epub 2018 Jan 4.
10
Long-term adverse event profile from four completed trials of oral eliglustat in adults with Gaucher disease type 1.来自四项已完成的口服 eliglustat 治疗 1 型戈谢病成人患者的长期不良事件概况。
Orphanet J Rare Dis. 2019 Jun 7;14(1):128. doi: 10.1186/s13023-019-1085-6.

引用本文的文献

1
Navigating gynaecological challenges in Gaucher's disease: insights from five European countries.应对戈谢病中的妇科挑战:来自五个欧洲国家的见解
Ther Adv Rare Dis. 2025 Apr 28;6:26330040251335665. doi: 10.1177/26330040251335665. eCollection 2025 Jan-Dec.
2
Women with Gaucher Disease.患有戈谢病的女性。
Biomedicines. 2024 Mar 5;12(3):579. doi: 10.3390/biomedicines12030579.
3
Long-Term Outcomes of Disease Modifying Therapies in Gaucher Disease.戈谢病中疾病修饰疗法的长期疗效

本文引用的文献

1
Effect of eliglustat on the pharmacokinetics of digoxin, metoprolol, and oral contraceptives and absorption of eliglustat when coadministered with acid-reducing agents.依利格鲁司他对地高辛、美托洛尔和口服避孕药药代动力学的影响,以及与抑酸剂合用对依利格鲁司他吸收的影响。
Mol Genet Metab. 2020 Apr;129(4):278-285. doi: 10.1016/j.ymgme.2020.01.001. Epub 2020 Jan 7.
2
Long-term adverse event profile from four completed trials of oral eliglustat in adults with Gaucher disease type 1.来自四项已完成的口服 eliglustat 治疗 1 型戈谢病成人患者的长期不良事件概况。
Orphanet J Rare Dis. 2019 Jun 7;14(1):128. doi: 10.1186/s13023-019-1085-6.
3
Indian J Pediatr. 2025 Jun;92(6):585-591. doi: 10.1007/s12098-023-04986-y. Epub 2024 Feb 5.
4
Long-term eliglustat treatment of Gaucher patients over up to 10 years in Vienna.维也纳长达 10 年的依鲁司他治疗戈谢病患者的长期疗效。
Wien Klin Wochenschr. 2022 Jun;134(11-12):471-477. doi: 10.1007/s00508-022-02021-2. Epub 2022 Apr 12.
Outcomes after 8 years of eliglustat therapy for Gaucher disease type 1: Final results from the Phase 2 trial.
依利格鲁司他治疗戈谢病 1 型 8 年的结果:来自 2 期试验的最终结果。
Am J Hematol. 2019 Jan;94(1):29-38. doi: 10.1002/ajh.25300. Epub 2018 Oct 26.
4
Once- versus twice-daily dosing of eliglustat in adults with Gaucher disease type 1: The Phase 3, randomized, double-blind EDGE trial.依利格鲁司他治疗 1 型戈谢病成人患者的每日一次与每日两次给药:III 期、随机、双盲 EDGE 试验。
Mol Genet Metab. 2018 Mar;123(3):347-356. doi: 10.1016/j.ymgme.2017.12.001. Epub 2018 Jan 4.
5
Outcomes after 18 months of eliglustat therapy in treatment-naïve adults with Gaucher disease type 1: The phase 3 ENGAGE trial.18个月依利格鲁司他治疗初治1型戈谢病成年患者的疗效:3期ENGAGE试验。
Am J Hematol. 2017 Nov;92(11):1170-1176. doi: 10.1002/ajh.24877. Epub 2017 Oct 3.
6
Eliglustat maintains long-term clinical stability in patients with Gaucher disease type 1 stabilized on enzyme therapy.在接受酶替代疗法病情已稳定的1型戈谢病患者中, eliglustat可维持长期临床稳定性。
Blood. 2017 Apr 27;129(17):2375-2383. doi: 10.1182/blood-2016-12-758409. Epub 2017 Feb 6.
7
Reported outcomes of 453 pregnancies in patients with Gaucher disease: An analysis from the Gaucher outcome survey.戈谢病患者453例妊娠的报告结局:来自戈谢病结局调查的分析
Blood Cells Mol Dis. 2018 Feb;68:226-231. doi: 10.1016/j.bcmd.2016.10.003. Epub 2016 Oct 20.
8
Eliglustat compared with imiglucerase in patients with Gaucher's disease type 1 stabilised on enzyme replacement therapy: a phase 3, randomised, open-label, non-inferiority trial.依利格鲁司他与伊米苷酶治疗稳定的 1 型戈谢病患者的比较:一项 3 期、随机、开放标签、非劣效性试验。
Lancet. 2015 Jun 13;385(9985):2355-62. doi: 10.1016/S0140-6736(14)61841-9. Epub 2015 Mar 26.
9
Effect of oral eliglustat on splenomegaly in patients with Gaucher disease type 1: the ENGAGE randomized clinical trial.口服依利格鲁司他对1型戈谢病患者脾肿大的影响:ENGAGE随机临床试验
JAMA. 2015 Feb 17;313(7):695-706. doi: 10.1001/jama.2015.459.
10
Eliglustat, an investigational oral therapy for Gaucher disease type 1: Phase 2 trial results after 4 years of treatment.依利格鲁司他,一种用于1型戈谢病的研究性口服疗法:4年治疗后的2期试验结果。
Blood Cells Mol Dis. 2014 Dec;53(4):274-6. doi: 10.1016/j.bcmd.2014.04.002. Epub 2014 May 15.