Key result
Novel MYBPC3 frameshift variant causing familial HCM drives allele-specific reduction of mutant transcripts.
Case Report
Identification of a novel MYBPC3 frameshift variant expands the spectrum of known genetic variants causing familial hypertrophic cardiomyopathy.
May inform variant interpretation in familial HCM families; leaves open mechanistic and therapeutic implications pending replication.
Hypertrophic cardiomyopathy (HCM) is the most common inherited heart disease with a prevalence of 1 : 200–1 : 500 in the general population. The majority of HCM-linked pathogenic (or likely pathogenic) variants is located in eight genes encoding proteins of sarcomere, the main contractile unit of cardiomyocytes; one of these genes, MYBPC3, is the most commonly affected and usually associated with the more benign clinical course of the disease compared to other HCM-related genes. Here, we describe a novel frame shift variant NM_000256.3:c.2781_2782insCACA of the MYBPC3 gene that causes familial HCM in the heterozygous state. The proband had a progressive heart failure despite the surgical removal of left ventricular tract obstruction. Evaluation of levels of transcripts produced from the mutant allele and wild-type allele of the MYBPC3 gene in proband myocardial tissue and comparison of their total levels with ones in the control samples from patients without HCM showed a significant allele-specific reduction of mutant transcript levels. Our results expand the spectrum of known genetic variants with a proven role in the development of HCM.
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Киселев et al. (2024) conducted a case report in Hypertrophic cardiomyopathy (HCM). MYBPC3 gene frameshift variant NM_000256.3:c.2781_2782insCACA vs. Control samples from patients without HCM was evaluated on Transcript levels in myocardial tissue. A novel MYBPC3 frameshift variant (NM_000256.3:c.2781_2782insCACA) causing familial hypertrophic cardiomyopathy resulted in a significant allele-specific reduction of mutant transcript levels.
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