A molecular screening strategy based on beta-myosin heavy chain, cardiac myosin binding protein C and troponin T genes in Italian patients with hypertrophic cardiomyopathy

J Cardiovasc Med (Hagerstown). 2006 Aug;7(8):601-7. doi: 10.2459/01.JCM.0000237908.26377.d6.

Abstract

Background: Mutations causing hypertrophic cardiomyopathy (HCM) have been described in nine different genes of the sarcomere. Three genes account for most known mutations: beta-myosin heavy chain (MYH7), cardiac myosin binding protein C (MYBPC3) and cardiac troponin T (TNNT2). Their prevalence in Italian HCM patients is unknown. Thus, we prospectively assessed a molecular screening strategy of these three genes in a consecutive population with HCM from two Italian centres.

Methods: Comprehensive screening of MYBPC3, MYH7 and TNNT2 was performed in 88 unrelated HCM patients by denaturing high-performance liquid chromatography and automatic sequencing.

Results: We identified 32 mutations in 50 patients (57%); 16 were novel. The prevalence rates for MYBPC3, MYH7 and TNNT2 were 32%, 17% and 2%, respectively. MYBPC3 mutations were 18, including two frameshift, five splice-site and two nonsense. All were 'private' except insC1065 and R502Q, present in three and two patients, respectively. Moreover, E258K was found in 14% of patients, suggesting a founder effect. MYH7 mutations were 12, all missense; seven were novel. In TNNT2, only two mutations were found. In addition, five patients had a complex genotype [i.e. carried a double MYBPC3 mutation (n = 2), or were double heterozygous for mutations in MYBPC3 and MYH7 (n = 3)].

Conclusions: The first comprehensive evaluation of MYBPC3, MYH7 and TNNT2 in an Italian HCM population allowed a genetic diagnosis in 57% of the patients. These data support a combined analysis of the three major sarcomeric genes as a rational and cost-effective initial approach to the molecular screening of HCM.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Cardiomyopathy, Hypertrophic / genetics*
  • Carrier Proteins / genetics*
  • Chi-Square Distribution
  • Chromatography, High Pressure Liquid
  • DNA Mutational Analysis
  • Female
  • Genotype
  • Humans
  • Italy
  • Male
  • Myosin Heavy Chains / genetics*
  • Pedigree
  • Phenotype
  • Polymerase Chain Reaction
  • Polymorphism, Genetic
  • Prevalence
  • Prospective Studies
  • Troponin T / genetics*

Substances

  • Carrier Proteins
  • Troponin T
  • myosin-binding protein C
  • Myosin Heavy Chains