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表现为急性单侧肾梗死的纤维肌性发育异常:一例病例报告及两种疾病综述

Fibromuscular Dysplasia Presenting as Acute Unilateral Renal Infarction: A Case Report and Review of Two Diseases.

作者信息

Abi Doumet Amanda, Bustos Brian, Garrell Jacob, Salman Momina, Haider Lalarukh

机构信息

Internal Medicine, University of Connecticut, Farmington, USA.

Primary Care Internal Medicine, University of Connecticut, Farmington, USA.

出版信息

Cureus. 2023 Mar 9;15(3):e35933. doi: 10.7759/cureus.35933. eCollection 2023 Mar.

DOI:10.7759/cureus.35933
PMID:37038580
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10082588/
Abstract

Fibromuscular dysplasia (FMD) is a rare systemic vascular disease that has been found to present as a renal infarction (RI) in only a handful of cases.  We present a case of a 53-year-old Vietnamese patient presenting for sharp, severe left-sided abdominal pain of two-day duration associated with a migraine headache. On presentation, she was afebrile, and her vital signs were stable. Laboratory investigations were significant for mildly elevated leukocytosis but were otherwise normal. CT abdomen and pelvis with contrast revealed a left-sided renal infarct. The patient was then admitted to the hospital and started on therapeutic anticoagulation. A transthoracic echocardiogram was obtained and revealed no vegetation. CT angiography of the abdomen was pursued and was significant for mild beading within the mid-right and left renal arteries, consistent with fibromuscular dysplasia. Our patient was diagnosed with renal infarction in the setting of fibromuscular dysplasia, a combination that has been reported only a few times. Interestingly, our patient also had mild FMD based on imaging, making it even more of an unusual cause of renal infarction. This case highlights the connection between these two diseases and the need for more studies to characterize the association between them.

摘要

纤维肌发育异常(FMD)是一种罕见的系统性血管疾病,仅在少数病例中被发现表现为肾梗死(RI)。我们报告一例53岁越南患者,因持续两天的剧烈左侧腹痛就诊,伴有偏头痛。就诊时,她无发热,生命体征稳定。实验室检查显示白细胞轻度升高,但其他方面正常。腹部和盆腔增强CT显示左侧肾梗死。该患者随后入院并开始接受抗凝治疗。进行了经胸超声心动图检查,未发现赘生物。随后进行了腹部CT血管造影,结果显示右肾动脉中段和左肾动脉轻度串珠样改变,符合纤维肌发育异常。我们的患者被诊断为纤维肌发育异常背景下的肾梗死,这种组合仅被报道过几次。有趣的是,根据影像学检查,我们的患者还患有轻度FMD,这使得它成为肾梗死更不寻常的病因。本病例突出了这两种疾病之间的联系以及开展更多研究以明确它们之间关联的必要性。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b09b/10082588/d0f645e7d09d/cureus-0015-00000035933-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b09b/10082588/17267ebd03d9/cureus-0015-00000035933-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b09b/10082588/d0f645e7d09d/cureus-0015-00000035933-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b09b/10082588/17267ebd03d9/cureus-0015-00000035933-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/b09b/10082588/d0f645e7d09d/cureus-0015-00000035933-i02.jpg

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本文引用的文献

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Current progress in clinical, molecular, and genetic aspects of adult fibromuscular dysplasia.成人纤维肌发育不良的临床、分子和遗传方面的最新进展。
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First International Consensus on the diagnosis and management of fibromuscular dysplasia.第一届国际纤维肌发育不良诊断与管理共识会议。
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Fibromuscular Dysplasia: Contemporary Concepts and Future Directions.纤维肌性发育不良:当代概念与未来方向。
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PHACTR1 Is a Genetic Susceptibility Locus for Fibromuscular Dysplasia Supporting Its Complex Genetic Pattern of Inheritance.PHACTR1是纤维肌发育不良的一个遗传易感性位点,支持其复杂的遗传模式。
PLoS Genet. 2016 Oct 28;12(10):e1006367. doi: 10.1371/journal.pgen.1006367. eCollection 2016 Oct.
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Acute renal artery occlusion: Presentation, treatment, and outcome.急性肾动脉闭塞:临床表现、治疗及预后
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Acute renal infarction resulting from fibromuscular dysplasia: a case report.纤维肌性发育异常导致的急性肾梗死:一例报告
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Intranuclear expression of progesterone receptors in smooth muscle cells of renovascular fibromuscular dysplasia: a pilot study.肾血管性纤维肌性发育异常平滑肌细胞中孕激素受体的核内表达:一项初步研究。
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Clinical manifestations of fibromuscular dysplasia vary by patient sex: a report of the United States registry for fibromuscular dysplasia.纤维肌性发育异常的临床表现因患者性别而异:美国纤维肌性发育异常登记处的报告
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