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妊娠或产褥期灾难性抗磷脂综合征的心肌受累:一例年轻哺乳期妇女病例及文献综述

Myocardial Involvement in Catastrophic Antiphospholipid Syndrome during Pregnancy or Puerperium: A Case of a Young Breastfeeding Woman and Literature Review.

作者信息

Varotto Leonardo, Spigolon Luca, Dotto Alberto, Leonardi Denis, Bragantini Giulia, Cerrito Luca Felice, Deluca Cristina, Hoxha Ariela

机构信息

Department of Cardiology, San Bortolo Hospital, viale Rodolfi 37, 36100 Vicenza, Italy.

Department of Radiology, San Bortolo Hospital, Viale Rodolfi 37, 36100 Vicenza, Italy.

出版信息

J Clin Med. 2024 Aug 12;13(16):4732. doi: 10.3390/jcm13164732.

DOI:10.3390/jcm13164732
PMID:39200874
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC11355751/
Abstract

Catastrophic Antiphospholipid Syndrome (CAPS) is a rare complication that can occur in patients with Antiphospholipid Syndrome (APS). CAPS occurs even more rarely during pregnancy/puerperium and pregnant patients, even less likely to show cardiac involvement without signs of damage on ultrasound and angiography with non-obstructive coronary arteries. We present a case of a 26-year-old breastfeeding woman, the youngest described with CAPS and acute myocardial infarction, whose diagnosis was made with cardiac magnetic resonance imaging (CMRI). A literature review of pregnant patients with similar problems was performed. There are diagnostic and therapeutic difficulties in treating these patients. CMRI demonstrated a transmural late enhancement area. A combination of therapies led to rapid clinical improvement. CMRI is an underused tool that reaffirms the pathophysiology of CAPS and leads clinicians to the possibility of a diffuse thrombotic process. CAPS involves more organs with high mortality rates. CMRI could be optimized in order to reach an early diagnosis and the most effective treatment. This study provides real-world evidence of the feasibility of MRI in a primary care setting during pregnancy/puerperium. Evidence from this study may influence future APS screening and inform policymakers regarding the use of leading MRI technology in the detection of the thrombotic process in a primary care setting.

摘要

灾难性抗磷脂综合征(CAPS)是抗磷脂综合征(APS)患者可能发生的一种罕见并发症。CAPS在妊娠/产褥期及孕妇中更为罕见,而孕妇出现心脏受累且超声和血管造影无损伤迹象、冠状动脉无阻塞的情况则更少见。我们报告一例26岁哺乳期女性病例,她是有记录以来最年轻的CAPS合并急性心肌梗死患者,其诊断通过心脏磁共振成像(CMRI)得以明确。我们对有类似问题的孕妇进行了文献综述。治疗这些患者存在诊断和治疗方面的困难。CMRI显示出透壁晚期强化区域。综合治疗使临床症状迅速改善。CMRI是一种未得到充分利用的工具,它再次证实了CAPS的病理生理学,并使临床医生认识到存在弥漫性血栓形成过程的可能性。CAPS累及更多器官,死亡率很高。可以对CMRI进行优化,以实现早期诊断和最有效的治疗。本研究提供了在妊娠/产褥期基层医疗环境中使用MRI可行性的真实世界证据。本研究的证据可能会影响未来APS的筛查,并为政策制定者提供关于在基层医疗环境中使用领先的MRI技术检测血栓形成过程的信息。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4117/11355751/159b6ef8da4c/jcm-13-04732-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4117/11355751/d668cf44defd/jcm-13-04732-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4117/11355751/159b6ef8da4c/jcm-13-04732-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4117/11355751/d668cf44defd/jcm-13-04732-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/4117/11355751/159b6ef8da4c/jcm-13-04732-g002.jpg

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本文引用的文献

1
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Semin Arthritis Rheum. 2024 Jun;66:152439. doi: 10.1016/j.semarthrit.2024.152439. Epub 2024 Mar 26.
2
Risk of fetal or neonatal death or neonatal intensive care unit admission associated with gadolinium magnetic resonance imaging exposure during pregnancy.妊娠期钆类磁共振成像暴露与胎儿或新生儿死亡或新生儿重症监护病房入院风险的关系。
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Prevalence, characteristics and outcome of cardiac manifestations in critically-ill antiphospholipid syndrome patients.
重症抗磷脂综合征患者心脏表现的患病率、特征及转归
J Autoimmun. 2022 Dec;133:102908. doi: 10.1016/j.jaut.2022.102908. Epub 2022 Sep 18.
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A catastrophic seronegative anti-phospholipid syndrome: case and literature review.一例灾难性血清阴性抗磷脂综合征:病例及文献综述
Thromb J. 2021 Dec 20;19(1):103. doi: 10.1186/s12959-021-00356-w.
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Catastrophic antiphospholipid syndrome presented as ruptured papillary muscle during puerperium in a patient with systemic lupus erythematosus.系统性红斑狼疮患者产后并发灾难性抗磷脂综合征致乳头肌破裂
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Combined brain/heart magnetic resonance imaging in antiphospholipid syndrome-two sides of the same coin.抗磷脂综合征的脑/心联合磁共振成像——同一疾病的两个方面。
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Combined Brain-Heart Magnetic Resonance Imaging in Autoimmune Rheumatic Disease Patients with Cardiac Symptoms: Hypothesis Generating Insights from a Cross-sectional Study.合并心脏症状的自身免疫性风湿病患者的脑心联合磁共振成像:一项横断面研究的假设生成性见解
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Complement activity and complement regulatory gene mutations are associated with thrombosis in APS and CAPS.补体活性和补体调节基因的突变与 APS 和 CAPS 中的血栓形成有关。
Blood. 2020 Jan 23;135(4):239-251. doi: 10.1182/blood.2019003863.
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Catastrophic antiphospholipid syndrome in pregnancy: a life-threatening condition.妊娠期灾难性抗磷脂综合征:一种危及生命的疾病。
BMJ Case Rep. 2019 Sep 16;12(9):e230863. doi: 10.1136/bcr-2019-230863.
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