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一位 25 岁沙特女性,患有抗合成酶综合征伴间质性肺病,病史 2 年,在妊娠第 3 个月末开始接受硫唑嘌呤治疗,并足月成功分娩。

A 25-Year-Old Saudi Woman with a 2-Year History of Antisynthetase Syndrome with Interstitial Lung Disease Who Commenced Azathioprine Treatment in the Third Trimester of Pregnancy and Had a Successful Birth at Term.

机构信息

Department of Obstetrics and Gynecology, King Saud University Medical City, Riyadh, Saudi Arabia.

出版信息

Am J Case Rep. 2022 Jul 7;23:e936833. doi: 10.12659/AJCR.936833.

DOI:10.12659/AJCR.936833
PMID:35794813
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC9274784/
Abstract

BACKGROUND Antisynthetase syndrome (ASS) is a rare systemic autoimmune disease. The clinical features of ASS include interstitial lung disease (ILD), myositis, arthritis, Raynaud's phenomenon, mechanic's hands, and unexplained fever. There is a paucity of reported cases and management guidelines in pregnancy. This report describes the case of a 25-year-old Saudi woman with a 2-year history of ASS with ILD who commenced azathioprine treatment in the third trimester and had a successful birth at term. CASE REPORT A 25-year-old Saudi primigravida woman with a 2-year history of ASS with ILD presented at 26 weeks of gestation after being lost to prepregnancy follow-up and discontinuing her medications. Azathioprine treatment was commenced, and despite poor prepregnancy follow-up, her pregnancy remained uneventful until 39 weeks, when fetal ultrasonography showed oligohydramnios. Therefore, labor induction was initiated, and she delivered vaginally with no postpartum complications or flare-ups. CONCLUSIONS The multisystem autoimmune disease ASS is a rare condition, and there are no clinical guidelines for its management in pregnant women. This case report highlights some aspects of ASS management and the importance of a multidisciplinary approach.

摘要

背景

抗合成酶综合征(ASS)是一种罕见的系统性自身免疫性疾病。ASS 的临床特征包括间质性肺病(ILD)、肌炎、关节炎、雷诺现象、技工手和不明原因的发热。在妊娠中,ASS 的报告病例和管理指南都很少。本报告描述了一例 25 岁沙特女性,患有 ASS 合并 ILD 病史 2 年,在妊娠晚期开始使用硫唑嘌呤治疗,并成功足月分娩。

病例报告

一位 25 岁的沙特初产妇,患有 ASS 合并 ILD,病史 2 年,在妊娠 26 周时因失去孕前随访和停止用药而就诊。开始使用硫唑嘌呤治疗,尽管孕前随访不佳,但她的妊娠仍顺利进行,直到 39 周时,胎儿超声检查发现羊水过少。因此,开始引产,她经阴道分娩,无产后并发症或病情复发。

结论

多系统自身免疫性疾病 ASS 较为罕见,目前尚无针对孕妇的临床管理指南。本病例报告强调了 ASS 管理的一些方面以及多学科方法的重要性。

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本文引用的文献

1
Antisynthetase Syndrome with Severe Interstitial Lung Disease in Pregnancy.妊娠合并严重间质性肺疾病的抗合成酶综合征
Case Rep Anesthesiol. 2021 Jul 26;2021:1150394. doi: 10.1155/2021/1150394. eCollection 2021.
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Maternal, foetal and child consequences of immunosuppressive drugs during pregnancy in women with organ transplant: a review.器官移植女性孕期使用免疫抑制药物对孕产妇、胎儿及儿童的影响:综述
Clin Kidney J. 2021 Mar 3;14(8):1871-1878. doi: 10.1093/ckj/sfab049. eCollection 2021 Aug.
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Antisynthetase syndrome - much more than just a myopathy.
抗合成酶综合征——远不止一种肌病。
Semin Arthritis Rheum. 2021 Feb;51(1):72-83. doi: 10.1016/j.semarthrit.2020.09.020. Epub 2020 Dec 22.
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Antisynthetase syndrome in pregnancy: A case and review of the literature.妊娠期抗合成酶综合征:1例病例及文献综述
Obstet Med. 2020 Jun;13(2):96-100. doi: 10.1177/1753495X18808646. Epub 2018 Nov 8.
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Clinical manifestations and treatment of antisynthetase syndrome.抗合成酶综合征的临床表现和治疗。
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[Antisynthetase syndromes].[抗合成酶综合征]
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Managing Crohn's Disease during Pregnancy.孕期克罗恩病的管理
Maedica (Bucur). 2016 Sep;11(3):221-226.
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New Insights into Antisynthetase Syndrome.抗合成酶综合征的新见解
Maedica (Bucur). 2016 Jun;11(2):130-135.
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The Diagnosis and Treatment of Antisynthetase Syndrome.抗合成酶综合征的诊断与治疗
Clin Pulm Med. 2016 Sep;23(5):218-226. doi: 10.1097/CPM.0000000000000171.
10
Pregnancy and autoimmune connective tissue diseases.妊娠与自身免疫性结缔组织病
Best Pract Res Clin Rheumatol. 2016 Feb;30(1):63-80. doi: 10.1016/j.berh.2016.05.002. Epub 2016 Jun 25.