Onorato Angela C, Gosselin Rachel, Chaudhari Bimal P, Alvarado Chance, White Peter, Garg Vidu, Bigelow Amee M
Res Sq. 2025 May 6:rs.3.rs-6314694. doi: 10.21203/rs.3.rs-6314694/v1.
Genetic testing guidelines for children in cardiac intensive care units (CICUs) are lacking despite a high prevalence of genetic diseases among this population. Advances in next-generation sequencing (NGS) technologies, especially exome and genome sequencing (ES/GS), enable a more comprehensive genetic evaluation than more traditional testing modalities such as chromosomal microarray (CMA). While testing recommendations exist for cardiomyopathies, primary arrhythmias, and pulmonary hypertension (PH), broad application of NGS, especially ES/GS, across indications for admission to CICUs has not been recommended. We aimed to evaluate the diagnostic efficacy of ES/GS in critically ill pediatric patients with cardiac disease via a retrospective chart review of patients who underwent clinical ES/GS in a quaternary hospital's pediatric CICU between January 2020 and August 2023. Forty-nine patients underwent ES/GS. Primary cardiac phenotypes included congenital heart disease, ventricular dysfunction, arrhythmia, and PH. Diagnostic results were found in 22 patients (44.9%) with 18/22 (81.8%) linked to cardiac phenotypes. Diagnostic yield was not different among primary cardiac phenotype groups but was higher in patients with ECA. CMA and gene panels would have failed to make a substantial proportion (80.6% and 36%, respectively) of diagnoses made by ES/GS. As NGS technologies and capabilities to interpret ES/GS data mature, diagnostic abilities in pediatric cardiac disease will continue to advance.
尽管心脏重症监护病房(CICU)中的儿童遗传疾病患病率很高,但针对该人群的基因检测指南却很缺乏。新一代测序(NGS)技术的进步,尤其是外显子组和基因组测序(ES/GS),能够比染色体微阵列(CMA)等更传统的检测方式进行更全面的基因评估。虽然针对心肌病、原发性心律失常和肺动脉高压(PH)已有检测建议,但尚未推荐在CICU入院指征中广泛应用NGS,尤其是ES/GS。我们旨在通过回顾性图表审查2020年1月至2023年8月在一家四级医院儿科CICU接受临床ES/GS检测的患者,评估ES/GS在患有心脏病的危重症儿科患者中的诊断效能。49名患者接受了ES/GS检测。主要心脏表型包括先天性心脏病、心室功能障碍、心律失常和PH。在22名患者(44.9%)中发现了诊断结果,其中18/22(81.8%)与心脏表型相关。主要心脏表型组之间的诊断率没有差异,但在患有ECA的患者中更高。CMA和基因检测板将无法做出ES/GS所做出的很大一部分诊断(分别为80.6%和36%)。随着NGS技术以及解读ES/GS数据能力的成熟,儿科心脏病的诊断能力将继续提高。