Mone F, Eberhardt, RY, et al. - Researchers aimed at investigating the incremental yield of antenatal exome sequencing (ES) over chromosomal microarray analysis (CMA) or conventional karyotyping in prenatally diagnosed congenital heart disease (CHD). They performed a prospective cohort study of 197 trios undergoing ES following CMA or karyotyping owing to prenatally diagnosed CHD and a systematic review of the literature. In this cohort, ES had an additional diagnostic yield of 12.7% in all CHD (25/197), 11.5% in isolated CHD (14/122) and 14.7% in CHD associated with ECA (11/75). The corresponding pooled incremental yields were 21%, 11% and 37%, respectively, from 18 studies (encompassing 636 CHD cases) included in the systematic review. This is the first systematic review performed on this topic. Overall findings suggested an apparent incremental yield of exome sequencing in these cases. Depending on the presence of robust pathways, consideration can be given to exome sequencing in prenatally diagnosed CHD , particularly for those with extracardiac abnormalities, while considerations may also be provided in cases that are isolated in nature.