儿科心脏通道疾病:最新遗传学资料。

IF 3 3区 医学 Q1 PEDIATRICS European Journal of Pediatrics Pub Date : 2024-11-01 Epub Date: 2024-09-23 DOI:10.1007/s00431-024-05757-3
Estefanía Martínez-Barrios, Oscar Campuzano, Andrea Greco, José Cruzalegui, Georgia Sarquella-Brugada
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引用次数: 0

摘要

心脏通道疾病是一组可导致恶性心律失常和心脏性猝死的遗传综合征,尤其是在儿童群体中。如今,12 导联心电图是诊断这些疾病最有效的工具。不完全渗透性和可变表达性是这些综合征的特征。其中一些恶性病变可能一直处于隐匿状态,只有运动、情绪或发热等诱因才能揭示心电模式,从而诊断疾病。心脏性猝死可能是这些综合征的首发症状。我们强烈建议使用能够早期诊断的辅助检查,其中包括:药理刺激、运动测试和基因分析。基因检测可以揭示疾病的起源,还能识别携带有害基因缺陷并因此处于危险中的家庭成员。该领域的主要挑战之一是大量意义不明的基因变异,这阻碍了将其有效转化为临床实践。及早识别儿科高危人群并进行适当的风险分层,对于采取个性化预防措施降低该人群的致死风险至关重要。已知信息:- 在儿科人群中,导致心脏性猝死的恶性心律失常主要由遗传综合征引起。- 确凿的基因诊断可揭示综合征的起源,并可进行级联筛查,以确定携带基因改变的亲属。最新进展- 测序技术的使用允许进行广泛的基因分析,有助于揭示导致遗传性心律失常综合征的新基因改变。- 定期对目前作用不明确的基因变异进行重新分析,将有助于识别真正致病的基因变异。
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Cardiac channelopathies in pediatrics: a genetic update.

Cardiac channelopathies are a group of inherited syndromes that can cause malignant arrhythmias and sudden cardiac death, particularly in the pediatric population. Today, a 12-lead electrocardiogram is the most effective tool to diagnose these diseases. Incomplete penetrance and variable expressivity are hallmarks of these syndromes. Some of these malignant entities may remain hidden and only a trigger such as exercise, emotions or fever can unmask the electrical pattern to diagnose the disease. Sudden cardiac death may be the first manifestation of any of these syndromes. The use of complementary tests that allow early diagnosis is strongly recommended, among which we find: pharmacological provocations, exercise tests, and genetic analysis. Genetic testing makes it possible to unravel the origin of the disease, and also identify family members who carry the harmful genetic defect and are therefore at risk. One of the main challenges in this area is the large number of genetic variants of uncertain significance, which prevent effective translation into clinical practice. Early identification of the pediatric population at risk and adequate risk stratification are crucial to adopting personalized preventive measures that reduce the risk of lethal episodes in this population. What is Known: • In the pediatric population, malignant arrhythmias leading to sudden cardiac death are mainly caused by inherited syndromes. • A conclusive genetic diagnosis unravels the origin of the syndrome and allows cascade screening to identify relatives carrying the genetic alteration. What is New: • The use of sequencing technologies allows a broad genetic analysis, helping to unravel new genetic alterations causing inherited arrhythmogenic syndromes. • A periodic reanalysis of genetic variants that currently have an ambiguous role will help discern those that are truly pathogenic.

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来源期刊
CiteScore
5.90
自引率
2.80%
发文量
367
审稿时长
3-6 weeks
期刊介绍: The European Journal of Pediatrics (EJPE) is a leading peer-reviewed medical journal which covers the entire field of pediatrics. The editors encourage authors to submit original articles, reviews, short communications, and correspondence on all relevant themes and topics. EJPE is particularly committed to the publication of articles on important new clinical research that will have an immediate impact on clinical pediatric practice. The editorial office very much welcomes ideas for publications, whether individual articles or article series, that fit this goal and is always willing to address inquiries from authors regarding potential submissions. Invited review articles on clinical pediatrics that provide comprehensive coverage of a subject of importance are also regularly commissioned. The short publication time reflects both the commitment of the editors and publishers and their passion for new developments in the field of pediatrics. EJPE is active on social media (@EurJPediatrics) and we invite you to participate. EJPE is the official journal of the European Academy of Paediatrics (EAP) and publishes guidelines and statements in cooperation with the EAP.
期刊最新文献
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