Key result
Exome sequencing reveals dual CASK and MYBPC3 variants driving a complex cardiomyopathy phenotype.
Why the study?
Whole-exome sequencing is increasingly used to study complex phenotypes where variants in more than one gene may cause disease, but its application in non-consanguineous families with complex cardiomyopathy phenotypes requires further evaluation.
Does whole-exome sequencing identify the genetic cause in a patient with a complex dilated cardiomyopathy phenotype?
Case Report (n=1)
No
Does whole-exome sequencing identify the genetic cause in a patient with a complex dilated cardiomyopathy phenotype?
Whole-exome sequencing can identify co-occurring rare genetic variants (such as in CASK and MYBPC3) responsible for complex phenotypes in non-consanguineous families.
Answers are grounded in this paper and cite their sources.
Broadens WES use to complex phenotypes; leaves open optimal indications and diagnostic yield in routine practice.
Whole-exome sequencing for clinical applications is now an integral part of medical genetics practice. Though most studies are performed in order to establish diagnoses in individuals with rare and clinically unrecognizable disorders, due to the constantly decreasing costs and commercial availability, whole-exome sequencing has gradually become the initial tool to study patients with clinically recognized disorders when more than one gene is responsible for the phenotype or in complex phenotypes, when variants in more than one gene can be the cause for the disease. Here we report a patient presenting with a complex phenotype consisting of severe, adult-onset, dilated cardiomyopathy, hearing loss and developmental delay, in which exome sequencing revealed two genetic variants that are inherited from a healthy mother: a novel missense variant in the CASK gene, mutations in which cause a spectrum of neurocognitive manifestations, and a second variant, in MYBPC3, that is associated with hereditary cardiomyopathy. We conclude that although the potential for co-occurrence of rare diseases is higher when analyzing undefined phenotypes in consanguineous families, it should also be given consideration in the genetic evaluation of complex phenotypes in non-consanguineous families.
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Reinstein et al. (2016) conducted a case report in Complex dilated cardiomyopathy phenotype (n=1). CASK and MYBPC3 mutations was evaluated on Genetic etiology of the complex phenotype. Exome sequencing identified that the patient's complex phenotype was caused by the co-occurrence of a novel missense variant in CASK and a known cardiomyopathy-associated variant in MYBPC3.
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