Cardiology Unit, Hospital Universitario San Ignacio, Bogotá, Colombia.
Faculty of Medicine, Pontificia Universidad Javeriana, Bogotá, Colombia.
ESC Heart Fail. 2023 Oct;10(5):3190-3194. doi: 10.1002/ehf2.14504. Epub 2023 Aug 26.
The case of a 49-year-old man with acute onset of heart failure is presented. The initial work-up showed a dilated cardiomyopathy with severely reduced left ventricular ejection fraction. In the differential diagnostic process, hypertensive, ischaemic, and valvular aetiologies were discarded. Subsequently, a cardiac magnetic resonance revealed global hypokinesis and inferior and anterior subepicardial fibrosis. Once differential diagnoses of subepicardial fibrosis (myocarditis, sarcoidosis, and Chagas disease) were discarded, a genetic panel was performed, resulting in a heterozygous mutation of desmoplakin (DSP) gene c.6697_6698del. A left-dominant DSP arrhythmogenic cardiomyopathy mutation was diagnosed. Structural myocardial abnormalities and ventricular arrhythmias characterize arrhythmogenic cardiomyopathy. Up to 50% of cases are associated with mutations in DSP genes (JUP, DSP, and PKP2). DSP is the fundamental component of the desmosome structure and provides structural support through intercellular adhesion. Therefore, when frequent differential diagnoses are discarded, genetic studies for dilated cardiomyopathy and DSP mutation should be considered.
现介绍 1 例 49 岁男性急性心力衰竭病例。初步检查显示扩张型心肌病伴严重左心室射血分数降低。在鉴别诊断过程中,排除了高血压、缺血性和瓣膜性病因。随后,心脏磁共振显示全心运动减弱,下壁和前壁心外膜下纤维化。排除心外膜下纤维化(心肌炎、结节病和恰加斯病)的鉴别诊断后,进行了基因检测,结果显示桥粒蛋白(DSP)基因 c.6697_6698del 存在杂合突变。诊断为左优势型 DSP 致心律失常性心肌病。结构心肌异常和室性心律失常是致心律失常性心肌病的特征。多达 50%的病例与 DSP 基因突变(JUP、DSP 和 PKP2)有关。DSP 是桥粒结构的基本组成部分,通过细胞间黏附提供结构支撑。因此,当频繁排除鉴别诊断时,应考虑对扩张型心肌病和 DSP 突变进行基因研究。