Key result
Cardiac targeted NGS identifies the genetic cause of severe antenatal cardiomyopathy in ~73% of families.
Why the study?
Severe cardiomyopathies observed during the antenatal period carry poor outcomes, but variable phenotypes and genetic heterogeneity make etiologic diagnosis difficult.
Observational (n=16)
Genetic testing using a targeted NGS panel has high diagnostic value for severe antenatal and early-onset cardiomyopathies, aiding in genetic counseling and identifying at-risk parents.
Cardiomyopathies are diseases of the heart muscle with variable clinical expressivity. Most of forms are inherited as dominant trait, and with incomplete penetrance until adulthood. Severe forms of cardiomyopathies were observed during the antenatal period with a pejorative issue leading to fetal death or medical interruption of pregnancy. Variable phenotypes and genetic heterogeneity make etiologic diagnosis difficult. We report 11 families (16 cases) whose unborn, newborn or infant with early onset cardiomyopathies. Detailed morphological and histological examinations of hearts were implemented, as well as genetic analysis on a cardiac targeted NGS panel. This strategy allowed the identification of the genetic cause of the cardiomyopathy in 8/11 families. Compound heterozygous mutations in dominant adulthood cardiomyopathy genes were found in two, pathogenic variants in co-dominant genes in one, de novo mutations in 5 including a germline mosaicism in one family. Parental testing was systematically performed to detect mutation carriers, and to manage cardiological surveillance and propose a genetic counseling. This study highlights the great diagnostic value of the genetic testing of severe antenatal cardiomyopathy both for genetic counseling and to detect presymptomatic parents at higher risk of developing cardiomyopathy.
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Kohaut et al. (2023) conducted an observational in Fetal cardiomyopathies (n=16). Cardiac targeted NGS panel was evaluated on Identification of the genetic cause of the cardiomyopathy. Genetic analysis using a cardiac targeted NGS panel identified the genetic cause of severe antenatal cardiomyopathy in 8 of 11 families (72.7%).
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