Key result
Whole-exome sequencing links a novel digenic mutation to severe LQTS in a Chinese family.
Case Report
The discovery of a digenic mutation in KCNH2 and JUP expands the spectrum of LQTS-related mutations and highlights the utility of whole-exome sequencing in the genetic diagnosis of malignant arrhythmias.
May prompt expanded sequencing in severe familial LQTS; hypothesis-generating and leaves open impact on risk stratification.
Long QT syndrome (LQTS), which is caused by an ion channel–related gene mutation, is a malignant heart disease with a clinical course of a high incidence of ventricular fibrillation and sudden cardiac death in the young. Mutations in KCNH2 (which encodes potassium voltage-gated channel subfamily H member 2) are responsible for LQTS in many patients. Here we report the novel mutation c.1898A>C in KCNH2 in a Chinese family with LQTS through whole-exome sequencing. The c.916dupA mutation in JUP (which encodes junction plakoglobin) is also discovered. Mutations in JUP were found to be associated with arrhythmogenic right ventricular cardiomyopathy. The double mutation in the proband may help explain his severe clinical manifestations, such as sudden cardiac death at an early age. Sequencing for the proband’s family members revealed that the KCNH2 mutation descends from his paternal line, while the mutation in JUP came from his maternal line. The data provided in this study may help expand the spectrum of LQTS-related KCNH2 mutations and add support to the genetic diagnosis and counseling of families affected by malignant arrhythmias.
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Zhai et al. (2020) conducted a case report in Long QT Syndrome. Digenic mutation (KCNH2 c.1898A>C and JUP c.916dupA) was evaluated. Whole-exome sequencing identified a novel digenic mutation (KCNH2 c.1898A>C and JUP c.916dupA) in a Chinese family with Long QT syndrome, potentially explaining severe clinical manifestations.
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