Watkins W Scott, Hernandez E Javier, Miller Thomas, Blue Nathan, Zimmerman Raquel, Griffiths Eric, Frise Erwin, Bernstein Daniel, Boskovski Marko, Brueckner Martina, Chung Wendy, Gaynor J William, Gelb Bruce, Goldmuntz Elizabeth, Gruber Peter, Newburger Jane, Roberts Amy, Morton Sarah, Mayer John, Seidman Christine, Seidman Jonathan, Shen Yufeng, Wagner Michael, Yost H Joseph, Yandell Mark, Tristani-Firouzi Martin
medRxiv. 2024 Nov 15:2024.05.03.24306784. doi: 10.1101/2024.05.03.24306784.
While exome and whole genome sequencing have transformed medicine by elucidating the genetic underpinnings of both rare and common complex disorders, its utility to predict clinical outcomes remains understudied. Here, we used artificial intelligence (AI) technologies to explore the predictive value of genome sequencing in forecasting clinical outcomes following surgery for congenital heart defects (CHD). We report results for a cohort of 2,253 CHD patients from the Pediatric Cardiac Genomics Consortium with a broad range of complex heart defects, pre- and post-operative clinical variables and exome sequencing. Damaging genotypes in chromatin-modifying and cilia-related genes were associated with an elevated risk of adverse post-operative outcomes, including mortality, cardiac arrest and prolonged mechanical ventilation. The impact of damaging genotypes was further amplified in the context of specific CHD phenotypes, surgical complexity and extra-cardiac anomalies. The absence of a damaging genotype in chromatin-modifying and cilia-related genes was also informative, reducing the risk for adverse postoperative outcomes. Thus, genome sequencing enriches the ability to forecast outcomes following congenital cardiac surgery.
虽然外显子组测序和全基因组测序通过阐明罕见和常见复杂疾病的遗传基础改变了医学,但它在预测临床结果方面的效用仍未得到充分研究。在这里,我们使用人工智能(AI)技术来探索基因组测序在预测先天性心脏病(CHD)手术后临床结果方面的预测价值。我们报告了来自儿科心脏基因组学联盟的2253名CHD患者队列的结果,这些患者患有广泛的复杂心脏缺陷、术前和术后临床变量以及外显子组测序。染色质修饰和纤毛相关基因中的有害基因型与术后不良结果的风险升高相关,包括死亡率、心脏骤停和延长机械通气时间。在特定的CHD表型、手术复杂性和心脏外异常的背景下,有害基因型的影响进一步放大。染色质修饰和纤毛相关基因中不存在有害基因型也具有参考价值,可降低术后不良结果的风险。因此,基因组测序增强了预测先天性心脏手术后结果的能力。