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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.

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

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