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病例报告:卡尼综合征的一个非典型病例。

Case Report: An Atypical Case of Carney Complex.

机构信息

Department of Medicine, University of Maryland School of Medicine, Baltimore, MD, USA.

Division of Cardiovascular Medicine, Department of Medicine, University of Maryland School of Medicine, Baltimore, MD, USA.

出版信息

Am J Case Rep. 2021 Oct 24;22:e933744. doi: 10.12659/AJCR.933744.

DOI:10.12659/AJCR.933744
PMID:34689149
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC8552419/
Abstract

BACKGROUND Intracardiac tumors are a rare entity, with myxomas being the most common among them (approximately 50% of intracardiac tumors). Up to 80% of myxomas originate within the left atrium and while most are incidental or isolated findings in asymptomatic patients, others may result in clinical manifestations of heart failure or emboli. Moreover, in some cases, myxomas can be part of a genetically inherited syndrome known as Carney complex (CNC), and present with varied phenotypes, including skin, endocrine, and neuroendocrine tumors. CASE REPORT We present a case of a 54-year-old male patient who presented with a several-month history of non-specific cough, dyspnea on exertion, and palpitations along with several skin tags, nevi, and nodules. He was found to have a retrocardiac density on chest X-ray, which was revealed to be a large left atrial myxoma on echocardiography. The myxoma was surgically excised and genetic testing for a mutation of the PRKAR1A gene (the most common mutation underlying CNC) was negative. However, 2 major clinical criteria for diagnosis of CNC were fulfilled based on cardiac myxoma and spotty skin pigmentation. In this report, we focus on the clinical manifestations of CNC, including guidance on tumor surveillance and genetic variants of CNC. CONCLUSIONS While CNC is most commonly associated with an inactivating mutation of the PRKAR1A gene, it can be diagnosed clinically in the absence of an identifiable genetic mutation. In patients presenting with atypical cardiac tumors, the early recognition of cutaneous manifestations can raise the index of suspicion for CNC, which can facilitate early diagnosis, treatment, and initiation of surveillance for neoplasia development.

摘要

背景

心脏内肿瘤较为罕见,其中黏液瘤最为常见(约占心脏内肿瘤的 50%)。多达 80%的黏液瘤起源于左心房,虽然大多数在无症状患者中为偶然或孤立发现,但其他患者可能出现心力衰竭或栓塞的临床表现。此外,在某些情况下,黏液瘤可能是一种称为 Carney 复合征(CNC)的遗传性综合征的一部分,表现出不同的表型,包括皮肤、内分泌和神经内分泌肿瘤。

病例报告

我们报告了一例 54 岁男性患者,其病史为数月的非特异性咳嗽、运动时呼吸困难和心悸,伴有多个皮肤标签、痣和结节。胸部 X 线检查发现心脏后密度,超声心动图显示为大型左心房黏液瘤。黏液瘤被手术切除,PRKAR1A 基因突变(CNC 最常见的突变)的基因检测为阴性。然而,根据心脏黏液瘤和斑片状皮肤色素沉着,满足 CNC 的 2 个主要临床诊断标准。在本报告中,我们重点介绍 CNC 的临床表现,包括肿瘤监测和 CNC 基因变异的指导。

结论

虽然 CNC 最常与 PRKAR1A 基因突变失活相关,但在没有可识别的基因突变的情况下也可以临床诊断。在出现非典型心脏肿瘤的患者中,早期识别皮肤表现可以提高 CNC 的怀疑指数,从而有助于早期诊断、治疗和开始监测肿瘤发展。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9591/8552419/39a429acad97/amjcaserep-22-e933744-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9591/8552419/b2636b44ed14/amjcaserep-22-e933744-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9591/8552419/2dc47bef459c/amjcaserep-22-e933744-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9591/8552419/f968b8d701ca/amjcaserep-22-e933744-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9591/8552419/39a429acad97/amjcaserep-22-e933744-g004.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9591/8552419/b2636b44ed14/amjcaserep-22-e933744-g001.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9591/8552419/2dc47bef459c/amjcaserep-22-e933744-g002.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9591/8552419/f968b8d701ca/amjcaserep-22-e933744-g003.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/9591/8552419/39a429acad97/amjcaserep-22-e933744-g004.jpg

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