Brugada Syndrome and Pulmonary Atresia with Intact Interventricular Septum: Fortuitous Finding or New Genetic Connection?

Genes (Basel). 2024 May 17;15(5):638. doi: 10.3390/genes15050638.

Abstract

Brugada syndrome is a rare arrhythmogenic syndrome associated mainly with pathogenic variants in the SCN5A gene. Right ventricle outflow tract fibrosis has been reported in some cases of patients diagnosed with Brugada syndrome. Pulmonary atresia with an intact ventricular septum is characterized by the lack of a functional pulmonary valve, due to the underdevelopment of the right ventricle outflow tract. We report, for the first time, a 4-year-old boy with pulmonary atresia with an intact ventricular septum who harbored a pathogenic de novo variant in SCN5A, and the ajmaline test unmasked a type-1 Brugada pattern. We suggest that deleterious variants in the SCN5A gene could be implicated in pulmonary atresia with an intact ventricular septum embryogenesis, leading to overlapping phenotypes.

Keywords: Brugada syndrome; genetics; pulmonary atresia; right ventricle outflow tract.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Brugada Syndrome* / genetics
  • Brugada Syndrome* / pathology
  • Child, Preschool
  • Heart Defects, Congenital / genetics
  • Heart Defects, Congenital / pathology
  • Humans
  • Male
  • NAV1.5 Voltage-Gated Sodium Channel* / genetics
  • Pulmonary Atresia* / genetics
  • Pulmonary Atresia* / pathology
  • Ventricular Septum / pathology

Substances

  • NAV1.5 Voltage-Gated Sodium Channel
  • SCN5A protein, human

Supplementary concepts

  • Pulmonary Atresia with Intact Ventricular Septum