Division of Pediatric Cardiology, Department of Pediatrics, Stanford University School of Medicine, 750 Welch Road, Suite 321, Palo Alto, CA, 94304, USA.
Lucile Packard Children's Hospital Stanford, Palo Alto, CA, USA.
Pediatr Cardiol. 2022 Mar;43(3):616-623. doi: 10.1007/s00246-021-02764-1. Epub 2021 Oct 29.
Hypertrophic cardiomyopathy (HCM) has historically been diagnosed phenotypically. Through genetic testing, identification of a molecular diagnosis (MolDx) is increasingly common but the impact on pediatric patients is unknown. This was a retrospective study of next-generation sequencing data for 602 pediatric patients with a clinician-reported history of HCM. Diagnostic yield was stratified by gene and self-reported race/ethnicity. A MolDx of HCM was identified in 242 (40%) individuals. Sarcomeric genes were the highest yielding, but pathogenic and/or likely pathogenic (P/LP) variants in syndromic genes were found in 36% of individuals with a MolDx, often in patients without documented clinical suspicion for a genetic syndrome. Among all MolDx, 73% were in genes with established clinical management recommendations and 2.9% were in genes that conferred eligibility for clinical trial enrollment. Black patients were the least likely to receive a MolDx. In the current era, genetic testing can impact management of HCM, beyond diagnostics or prognostics, through disease-specific guidelines or clinical trial eligibility. Genetic testing frequently can help identify syndromes in patients for whom syndromes may not be suspected. These findings highlight the importance of pursuing broad genetic testing, independent of suspicion based on phenotype. Lower rates of MolDx in Black patients may contribute to health inequities. Further research is needed evaluating the genetics of HCM in underrepresented/underserved populations. Additionally, research related to the impact of genetic testing on clinical management of other diseases is warranted.
肥厚型心肌病(HCM)的诊断历来是基于表型。通过基因检测,越来越多的患者可以确定分子诊断(MolDx),但这对儿科患者的影响尚不清楚。这是一项对 602 名儿科患者的下一代测序数据进行的回顾性研究,这些患者均有临床医生报告的 HCM 病史。根据基因和自我报告的种族/族裔,对诊断率进行了分层。在 242 名(40%)个体中确定了 HCM 的 MolDx。肌节基因的检出率最高,但在 MolDx 个体中,36%的个体存在综合征基因的致病性和/或可能致病性(P/LP)变异,这些个体通常没有遗传综合征的临床可疑记录。在所有 MolDx 中,73%的变异位于具有明确临床管理建议的基因中,2.9%的变异位于可使个体有资格参加临床试验的基因中。黑人患者最不可能接受 MolDx。在当前时代,基因检测除了诊断或预后外,还可以通过疾病特异性指南或临床试验资格,影响 HCM 的管理。基因检测通常可以帮助识别那些可能未被怀疑患有综合征的患者的综合征。这些发现强调了无论是否基于表型怀疑,均应广泛进行基因检测的重要性。黑人患者 MolDx 率较低可能导致健康不平等。需要进一步研究评估代表性不足/服务不足人群的 HCM 遗传学。此外,还需要研究基因检测对其他疾病临床管理的影响。