Cardiovascular Genetics Center, Institut d'Investigació Biomèdica de Girona (IDIBGI-CERCA), Parc Hospitalari Martí i Julià, Edifici M2, 17190 Salt, Spain.
Centro de Investigación Biomédica en Red Enfermedades Cardiovasculares (CIBERCV), 28029 Madrid, Spain.
Int J Mol Sci. 2024 Mar 29;25(7):3807. doi: 10.3390/ijms25073807.
Dilated cardiomyopathy is a heterogeneous entity that leads to heart failure and malignant arrhythmias. Nearly 50% of cases are inherited; therefore, genetic analysis is crucial to unravel the cause and for the early identification of carriers at risk. A large number of variants remain classified as ambiguous, impeding an actionable clinical translation. Our goal was to perform a comprehensive update of variants previously classified with an ambiguous role, applying a new algorithm of already available tools. In a cohort of 65 cases diagnosed with dilated cardiomyopathy, a total of 125 genetic variants were classified as ambiguous. Our reanalysis resulted in the reclassification of 12% of variants from an unknown to likely benign or likely pathogenic role, due to improved population frequencies. For all the remaining ambiguous variants, we used our algorithm; 60.9% showed a potential but not confirmed deleterious role, and 24.5% showed a potential benign role. Periodically updating the population frequencies is a cheap and fast action, making it possible to clarify the role of ambiguous variants. Here, we perform a comprehensive reanalysis to help to clarify the role of most of ambiguous variants. Our specific algorithms facilitate genetic interpretation in dilated cardiomyopathy.
扩张型心肌病是一种导致心力衰竭和恶性心律失常的异质性疾病。近 50%的病例是遗传性的;因此,基因分析对于揭示病因和早期识别有风险的携带者至关重要。大量变异仍被归类为不明确,阻碍了可采取行动的临床转化。我们的目标是应用已经可用的工具的新算法,对以前分类为不明确作用的变异进行全面更新。在一组诊断为扩张型心肌病的 65 例病例中,共有 125 种遗传变异被归类为不明确。由于人群频率的改善,我们的重新分析导致 12%的变异从未知到可能良性或可能致病性的作用进行了重新分类。对于所有剩余的不明确变体,我们使用了我们的算法;60.9%显示出潜在但未经证实的有害作用,24.5%显示出潜在的良性作用。定期更新人群频率是一种廉价且快速的行动,可以澄清不明确变体的作用。在这里,我们进行了全面的重新分析,以帮助澄清大多数不明确变体的作用。我们的特定算法有助于扩张型心肌病的遗传解释。