Suppr超能文献

基因型对肥厚型心肌病患者表型和预后的影响。

The influence of genotype on the phenotype and prognosis of patients with hypertrophic cardiomyopathy.

作者信息

Conde Inês Macedo, Grácio Machado Joana, Dias Mónica, Vieira Catarina, Pereira Vitor Hugo

机构信息

Cardiology Department, Hospital de Braga, Braga, Portugal.

São Victor, Rua das Sete Fontes, Braga, 4710-243, Portugal.

出版信息

Int J Cardiovasc Imaging. 2025 Sep 18. doi: 10.1007/s10554-025-03518-3.

Abstract

INTRODUCTION

Hypertrophic Cardiomyopathy (HCM) is a genetic Heart disease characterized by myocardial hypertrophy, which can lead to Heart failure and fatal arrhythmias. The genetic basis of this condition is complex, and its relationship with phenotype and prognosis is not completely understood. Our aim was to evaluate the influence of genotype on clinical and imaging phenotypes, and on a 2-year prognosis in HCM patients.Methods We included 117 HCM patients diagnosed by cardiac magnetic resonance (CMR) between 2018 and 2024. Only 77 patients in the cohort underwent genetic testing and were included in the genetic analysis. Genetic testing results were classified as negative, variants of uncertain significance (VUS), or pathogenic, and correlated with clinical and imaging data. For prognosis, we analyzed major adverse cardiovascular events (MACE) and hospitalizations over 2 years.

RESULTS

Genetic mutations (pathogenic or VUS) were identified in 55.9% of patients and were associated with greater symptom burden (p = 0.021). Both pathogenic and VUS carriers were more symptomatic than mutation-negative patients (p = 0.049 and p = 0.041), with no significant differences between the two groups. Patients with mutations also showed higher prevalence of LGE (p = 0.013), particularly those with pathogenic variants (p = 0.002). Compared to mutation-negative patients, both pathogenic and VUS carriers had more LGE (p = 0.007 and p = 0.005), while pathogenic variant carriers additionally demonstrated greater LV wall thickness (p = 0.042). During follow-up, pathogenic variant carriers had increased risk of MACE (HR 4.40, p < 0.001) and cardiovascular hospitalizations (HR 3.24, p = 0.033) versus mutation-negative patients. Family history of HCM or sudden cardiac death was associated with higher MACE risk (HR 2.33, p = 0.017; HR 3.23, p = 0.001). Patients with thin filament mutations, particularly TPM1, presented with worse imaging phenotypes and higher incidence of non-sustained ventricular tachycardia (80% vs. 0% in mutation-negative).

CONCLUSION

Genotype influences clinical and imaging phenotypes and short-term outcomes in HCM patients, underscoring the importance of genetic testing in risk stratification. However, small sample size limits conclusions about specific gene variants.

摘要

引言

肥厚型心肌病(HCM)是一种遗传性心脏病,其特征为心肌肥厚,可导致心力衰竭和致命性心律失常。这种疾病的遗传基础复杂,其与表型和预后的关系尚未完全明确。我们的目的是评估基因型对HCM患者临床和影像学表型以及2年预后的影响。

方法

我们纳入了2018年至2024年间经心脏磁共振成像(CMR)诊断的117例HCM患者。队列中只有77例患者接受了基因检测并纳入基因分析。基因检测结果分为阴性、意义未明变异(VUS)或致病性,并与临床和影像学数据相关联。对于预后,我们分析了2年内的主要不良心血管事件(MACE)和住院情况。

结果

55.9%的患者检测到基因突变(致病性或VUS),且与更重的症状负担相关(p = 0.021)。致病性和VUS携带者均比无突变患者症状更明显(p = 0.049和p = 0.041),两组间无显著差异。有突变的患者LGE患病率也更高(p = 0.013),尤其是那些携带致病性变异的患者(p = 0.002)。与无突变患者相比,致病性和VUS携带者的LGE均更多(p = 0.007和p = 0.005),而致病性变异携带者的左心室壁厚度更大(p = 0.042)。随访期间,与无突变患者相比,致病性变异携带者发生MACE的风险增加(HR 4.40,p < 0.001)以及心血管住院风险增加(HR 3.24,p = 0.033)。HCM家族史或心源性猝死家族史与更高的MACE风险相关(HR 2.33,p = 0.017;HR 3.23,p = 0.001)。携带细肌丝基因突变(尤其是TPM1)的患者影像学表型更差,非持续性室性心动过速发生率更高(无突变患者为0%,携带者为80%)。

结论

基因型影响HCM患者的临床和影像学表型以及短期预后,强调了基因检测在风险分层中的重要性。然而,样本量较小限制了关于特定基因变异结论的得出。

文献检索

告别复杂PubMed语法,用中文像聊天一样搜索,搜遍4000万医学文献。AI智能推荐,让科研检索更轻松。

立即免费搜索

文件翻译

保留排版,准确专业,支持PDF/Word/PPT等文件格式,支持 12+语言互译。

免费翻译文档

深度研究

AI帮你快速写综述,25分钟生成高质量综述,智能提取关键信息,辅助科研写作。

立即免费体验