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与核纤层蛋白A/C基因第11外显子突变相关的复杂表型:肥厚型心肌病、房室传导阻滞、严重血脂异常和糖尿病。

Complex phenotype linked to a mutation in exon 11 of the lamin A/C gene: Hypertrophic cardiomyopathy, atrioventricular block, severe dyslipidemia and diabetes.

作者信息

Francisco Ana Rita G, Santos Gonçalves Inês, Veiga Fátima, Mendes Pedro Mónica, Pinto Fausto J, Brito Dulce

机构信息

Cardiology Department, Santa Maria University Hospital, CHLN, CAML, CCUL, Faculty of Medicine, University of Lisbon, Lisbon, Portugal.

Cardiology Department, Santa Maria University Hospital, CHLN, CAML, CCUL, Faculty of Medicine, University of Lisbon, Lisbon, Portugal.

出版信息

Rev Port Cardiol. 2017 Sep;36(9):669.e1-669.e4. doi: 10.1016/j.repc.2016.07.018. Epub 2017 Sep 3.

Abstract

The lamin A/C (LMNA) gene encodes lamins A and C, which have an important role in nuclear cohesion and chromatin organization. Mutations in this gene usually lead to the so-called laminopathies, the primary cardiac manifestations of which are dilated cardiomyopathy and intracardiac conduction defects. Some mutations, associated with lipodystrophy but not cardiomyopathy, have been linked to metabolic abnormalities such as diabetes and severe dyslipidemia. Herein we describe a new phenotype associated with a mutation in exon 11 of the LMNA gene: hypertrophic cardiomyopathy, atrioventricular block, severe dyslipidemia and diabetes. A 64-year-old woman with hypertrophic cardiomyopathy and a point mutation in exon 11 of the LMNA gene (c.1718C>T, Ser573Leu) presented with severe symptomatic ventricular hypertrophy and left ventricular outflow tract obstruction. She underwent septal alcohol ablation, followed by Morrow myectomy. The patient was also diagnosed with severe dyslipidemia, diabetes and obesity, and fulfilled diagnostic criteria for metabolic syndrome. No other characteristics of LMNA mutation-related phenotypes were identified. The development of type III atrioventricular block with no apparent cause, and mildly depressed systolic function, prompted referral for cardiac resynchronization therapy. In conclusion, the association between LMNA mutations and different phenotypes is complex and not fully understood, and can present with a broad spectrum of severity.

摘要

核纤层蛋白A/C(LMNA)基因编码核纤层蛋白A和C,它们在核内聚合力和染色质组织中起重要作用。该基因的突变通常会导致所谓的核纤层蛋白病,其主要心脏表现为扩张型心肌病和心内传导缺陷。一些与脂肪营养不良而非心肌病相关的突变与糖尿病和严重血脂异常等代谢异常有关。在此,我们描述了一种与LMNA基因第11外显子突变相关的新表型:肥厚型心肌病、房室传导阻滞、严重血脂异常和糖尿病。一名64岁患有肥厚型心肌病且LMNA基因第11外显子存在点突变(c.1718C>T,Ser573Leu)的女性,出现严重的有症状性心室肥厚和左心室流出道梗阻。她接受了酒精间隔消融术,随后进行了Morrow心肌切除术。该患者还被诊断出患有严重血脂异常、糖尿病和肥胖症,并符合代谢综合征的诊断标准。未发现与LMNA突变相关表型的其他特征。无明显原因出现III度房室传导阻滞以及轻度收缩功能减退,促使患者接受心脏再同步治疗。总之,LMNA突变与不同表型之间的关联复杂且尚未完全明确,其严重程度范围广泛。

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