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经心内膜心肌活检证实的类风湿关节炎患者应用羟氯喹所致心肌病。

Endomyocardial biopsy-proven hydroxychloroquine-induced cardiomyopathy in a patient with rheumatoid arthritis.

机构信息

Department of Internal Medicine, University of Kansas Medical Center, Kansas City, Kansas, USA.

Department of Cardiovascular Medicine, University of Kansas Medical Center, Kansas City, Kansas, USA.

出版信息

BMJ Case Rep. 2023 Apr 13;16(4):e252327. doi: 10.1136/bcr-2022-252327.

DOI:10.1136/bcr-2022-252327
PMID:37055077
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10105987/
Abstract

Hydroxychloroquine is a disease-modifying antirheumatic drug used for various rheumatological conditions. Its long-term use is well-known to have toxic effects on cardiac muscle cells. We present a biopsy-proven case of hydroxychloroquine-induced cardiotoxicity with detailed histopathological and imaging findings. The patient was referred to our heart failure clinic for concerns of reduction in left ventricular ejection fraction despite being on guideline-directed medical therapy. She had been diagnosed with rheumatoid arthritis, pulmonary hypertension and then subsequently heart failure with reduced ejection fraction 5 years ago. The evaluation included right heart catheterisation, cardiac MRI and endomyocardial biopsy. Light and electron microscopy showed myocyte hypertrophy and vacuolar change, abnormal mitochondria, myeloid bodies and curvilinear bodies. These findings were specific for hydroxychloroquine-induced cardiomyopathy. This case highlights the importance of clinical monitoring, early suspicion and consideration of drug-induced toxicities as a culprit for heart failure.

摘要

羟氯喹是一种用于各种风湿性疾病的疾病修饰抗风湿药物。众所周知,长期使用会对心肌细胞产生毒性作用。我们报告了一例经活检证实的羟氯喹诱导性心肌病病例,详细描述了其组织病理学和影像学表现。该患者因左心室射血分数降低而被转诊到我们的心力衰竭诊所,尽管她正在接受指南指导的药物治疗。她 5 年前被诊断患有类风湿关节炎、肺动脉高压,随后又被诊断为射血分数降低的心力衰竭。评估包括右心导管检查、心脏 MRI 和心内膜心肌活检。光镜和电镜显示心肌细胞肥大和空泡化、线粒体异常、髓样小体和曲线形小体。这些发现与羟氯喹诱导性心肌病相符。该病例强调了临床监测、早期怀疑和考虑药物诱导的毒性作为心力衰竭病因的重要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b1b9/10105987/f249e373dce9/bcr-2022-252327f03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b1b9/10105987/1412b7dcc11f/bcr-2022-252327f01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b1b9/10105987/5a9c6085f5d8/bcr-2022-252327f02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b1b9/10105987/f249e373dce9/bcr-2022-252327f03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b1b9/10105987/1412b7dcc11f/bcr-2022-252327f01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b1b9/10105987/5a9c6085f5d8/bcr-2022-252327f02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b1b9/10105987/f249e373dce9/bcr-2022-252327f03.jpg

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本文引用的文献

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Hydroxychloroquine: a comprehensive review and its controversial role in coronavirus disease 2019.羟氯喹:全面综述及其在 2019 冠状病毒病中的争议性作用。
Ann Med. 2021 Dec;53(1):117-134. doi: 10.1080/07853890.2020.1839959.
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Ultrastructural deposits appearing as "zebra bodies" in renal biopsy: Fabry disease?- comparative case reports.肾活检中出现“斑马小体”的超微结构沉积物:法布里病?——病例对照报告
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Hydroxychloroquine cardiotoxicity presenting as a rapidly evolving biventricular cardiomyopathy: key diagnostic features and literature review.羟氯喹致心脏毒性表现为快速进展性双心室心肌病:关键诊断特征及文献复习。
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