Congenital polyvalvular disease expands the cardiac phenotype of the RASopathies

Am J Med Genet A. 2021 May;185(5):1486-1493. doi: 10.1002/ajmg.a.62146. Epub 2021 Mar 8.

Abstract

The RASopathies are a group of similar genetic syndromes with cardiovascular abnormalities, characteristic facial features, short stature, abnormalities of the skin and musculoskeletal system, and variable neurodevelopmental challenges. The most common cardiovascular abnormalities include pulmonary valvular stenosis and hypertrophic cardiomyopathy. Congenital polyvalvular disease (CPVD) refers to congenital dysplasia of two or more cardiac valves. We diagnosed a RASopathy in two individuals with CPVD and noted that CPVD in RASopathies has rarely been reported in the literature. Thus, we performed a retrospective chart review and literature review to investigate the association and characterize the phenotype of CPVD in the RASopathies. CPVD was present in 2.5% (n = 6/243) of individuals in our RASopathy cohort. Involvement of two cardiac valves, commonly the aortic and pulmonic valves, was seen in the majority of individuals (6/8; 75%) in our cohort, but only 27% (3/11) of reported CPVD and RASopathy cases in the literature. CPVD should be considered an associated cardiovascular phenotype of the RASopathies, which has implications for diagnosis and management.

Keywords: Noonan syndrome; PTPN11; RASopathies; cardiofaciocutaneous (CFC) syndrome; connective tissue disease; polyvalvular disease.

Publication types

  • Research Support, N.I.H., Extramural
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Aortic Valve / pathology
  • Cardiomyopathy, Hypertrophic / epidemiology
  • Cardiomyopathy, Hypertrophic / genetics*
  • Cardiomyopathy, Hypertrophic / pathology
  • Cardiovascular Abnormalities / epidemiology
  • Cardiovascular Abnormalities / genetics
  • Cardiovascular Abnormalities / pathology
  • Child
  • Child, Preschool
  • Dwarfism / genetics
  • Dwarfism / pathology
  • Facies
  • Female
  • Heart Defects, Congenital / genetics
  • Heart Defects, Congenital / pathology
  • Humans
  • Infant
  • Infant, Newborn
  • Male
  • Musculoskeletal Abnormalities / epidemiology
  • Musculoskeletal Abnormalities / genetics
  • Musculoskeletal Abnormalities / pathology
  • Noonan Syndrome
  • Phenotype
  • Protein Tyrosine Phosphatase, Non-Receptor Type 11 / genetics*
  • Proto-Oncogene Proteins B-raf / genetics*
  • Pulmonary Valve Stenosis / epidemiology
  • Pulmonary Valve Stenosis / genetics*
  • Pulmonary Valve Stenosis / pathology
  • Skin Abnormalities / genetics
  • Skin Abnormalities / pathology
  • ras Proteins / genetics

Substances

  • BRAF protein, human
  • Proto-Oncogene Proteins B-raf
  • PTPN11 protein, human
  • Protein Tyrosine Phosphatase, Non-Receptor Type 11
  • ras Proteins