Whole blood RNA offers a rapid, comprehensive approach to genetic diagnosis of cardiovascular diseases

Genet Med. 2007 Jan;9(1):23-33. doi: 10.1097/gim.0b013e31802d74de.

Abstract

Purpose: Long QT Syndrome, Marfan Syndrome, hypertrophic and dilated cardiomyopathy are caused by mutations in large, multi-exon genes that are principally expressed in cardiovascular tissues. Genetic testing for these disorders is labor-intensive and expensive. We sought to develop a more rapid, comprehensive, and cost-effective approach.

Methods: Paired whole blood samples were collected into tubes with or without an RNA-preserving solution, and harvested for whole blood RNA or leukocyte DNA, respectively. Large overlapping cDNA fragments from KCNQ1 and KCNH2 (Long QT Syndrome), MYBPC3 (hypertrophic and dilated cardiomyopathy), or FBN1 (Marfan Syndrome) were amplified from RNA and directly sequenced. Variants were confirmed in leukocyte DNA.

Results: All 4 transcripts were amplified and sequenced from whole blood mRNA. Six known and 2 novel mutations were first identified from RNA of 10 probands, and later confirmed in genomic DNA, at considerable savings in time and cost. In one patient with MFS, RNA sequencing directly identified a splicing mutation. Results from RNA and DNA were concordant for single nucleotide polymorphisms at the same loci.

Conclusion: Taking advantage of new whole blood RNA stabilization methods, we have designed a cost-effective, comprehensive method for mutation detection that should significantly facilitate clinical genetic testing in four lethal cardiovascular disorders.

Publication types

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

MeSH terms

  • Adolescent
  • Adult
  • Cardiomyopathy, Dilated / blood
  • Cardiomyopathy, Dilated / diagnosis
  • Cardiomyopathy, Dilated / genetics
  • Cardiomyopathy, Hypertrophic / blood
  • Cardiomyopathy, Hypertrophic / diagnosis
  • Cardiomyopathy, Hypertrophic / genetics
  • Cardiovascular Diseases / blood
  • Cardiovascular Diseases / diagnosis*
  • Cardiovascular Diseases / genetics
  • Carrier Proteins / genetics*
  • Child
  • ERG1 Potassium Channel
  • Ether-A-Go-Go Potassium Channels / genetics*
  • Exons / genetics
  • Female
  • Fibrillin-1
  • Fibrillins
  • Genetic Testing
  • Genotype
  • Humans
  • Infant
  • Infant, Newborn
  • KCNQ1 Potassium Channel / genetics*
  • Long QT Syndrome / blood
  • Long QT Syndrome / diagnosis
  • Long QT Syndrome / genetics
  • Male
  • Marfan Syndrome / blood
  • Marfan Syndrome / diagnosis
  • Marfan Syndrome / genetics
  • Microfilament Proteins / genetics*
  • Middle Aged
  • Mutation / genetics
  • Polymorphism, Single Nucleotide / genetics*
  • Prospective Studies
  • RNA / genetics
  • RNA / metabolism
  • Reverse Transcriptase Polymerase Chain Reaction

Substances

  • Carrier Proteins
  • ERG1 Potassium Channel
  • Ether-A-Go-Go Potassium Channels
  • FBN1 protein, human
  • Fibrillin-1
  • Fibrillins
  • KCNH2 protein, human
  • KCNQ1 Potassium Channel
  • Microfilament Proteins
  • myosin-binding protein C
  • RNA