Identification of a novel de novo deletion in RAF1 associated with biventricular hypertrophy in Noonan syndrome

Am J Med Genet A. 2014 Aug;164A(8):2069-73. doi: 10.1002/ajmg.a.36588. Epub 2014 Apr 29.

Abstract

Biventricular hypertrophy (BVH) is a disease state characterized by the thickening of the ventricle walls. The differential diagnosis of BVH with other congenital and familial diseases in which increased ventricle wall thickness is a prominent clinical feature is fundamental due to its therapeutic and prognostic value, mainly during infancy. We describe a 2-month-old infant presenting BVH. Using exome sequencing, we identified a novel de novo 3-bp deletion in the RAF1 gene that is located in the binding active site for the 14-3-3 peptide. Based on docking calculations, we demonstrate that this novel mutation impairs protein/target binding, thus constitutively activating Ras signaling, which is a dysregulation associated with Noonan syndrome. Finally, our study underlines the importance of molecular modeling to understand the roles of novel mutations in pathogenesis.

Keywords: 14-3-3 protein; Noonan syndrome; RAF1; biventricular hypertrophy; computational modeling; exome sequencing.

Publication types

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

MeSH terms

  • 14-3-3 Proteins / chemistry
  • 14-3-3 Proteins / metabolism
  • Adult
  • Binding Sites
  • Cardiomegaly / diagnosis
  • Cardiomegaly / genetics*
  • DNA Mutational Analysis
  • Exome
  • Female
  • Gene Deletion*
  • Genetic Association Studies*
  • Humans
  • Infant
  • Male
  • Models, Molecular
  • Mutation
  • Noonan Syndrome / diagnosis
  • Noonan Syndrome / genetics*
  • Protein Binding
  • Protein Conformation
  • Proto-Oncogene Proteins c-raf / chemistry
  • Proto-Oncogene Proteins c-raf / genetics*
  • Proto-Oncogene Proteins c-raf / metabolism

Substances

  • 14-3-3 Proteins
  • Proto-Oncogene Proteins c-raf