Arrhythmogenic right ventricular dysplasia/cardiomyopathy diagnostic task force criteria: impact of new task force criteria

Circ Arrhythm Electrophysiol. 2010 Apr;3(2):126-33. doi: 10.1161/CIRCEP.109.927202. Epub 2010 Mar 9.

Abstract

Background: Arrhythmogenic Right Ventricular Dysplasia/Cardiomyopathy (ARVD/C) Diagnostic Task Force Criteria (TFC) proposed in 1994 are highly specific but lack sensitivity. A new international task force modified criteria to improve diagnostic yield. A comparison of diagnosis by 1994 TFC versus newly proposed criteria in 3 patient groups was conducted.

Methods and results: In new TFC, scoring by major and minor criteria is maintained. Structural abnormalities are quantified and TFC highly specific for ARVD/C upgraded to major. Furthermore, new criteria are added: terminal activation duration of QRS > or = 55 ms, ventricular tachycardia with left bundle-branch block morphology and superior axis, and genetic criteria. Three groups were studied: (1) 105 patients with proven ARVD/C according to 1994 TFC, (2) 89 of their family members, and (3) 39 patients with probable ARVD/C (ie, 3 points by 1994 TFC). All were screened for pathogenic mutations in desmosomal genes. Three ARVD/C patients did not meet the new sharpened criteria on structural abnormalities and thereby did not fulfill new TFC. In 62 of 105 patients with proven ARVD/C, mutations were found: 58 in the gene encoding Plakophilin2 (PKP2), 3 in Desmoglein2, 3 in Desmocollin2, and 1 in Desmoplakin. Three patients had bigenic involvement. Ten additional relatives (11%) fulfilled new TFC: 9 (90%) were female, and all carried PKP2 mutations. No relatives lost diagnosis by application of new TFC. Of patients with probable ARVD/C, 25 (64%) fulfilled new TFC: 8 (40%) women and 14 (56%) carrying pathogenic mutations.

Conclusions: In this first study applying new TFC to patients suspected of ARVD/C, 64% of probable ARVD/C patients and 11% of family members were additionally diagnosed. ECG criteria and pathogenic mutations especially contributed to new diagnosis. Newly proposed TFC have a major impact in increasing diagnostic yield of ARVD/C.

Publication types

  • Comparative Study
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adult
  • Arrhythmogenic Right Ventricular Dysplasia / diagnosis*
  • Arrhythmogenic Right Ventricular Dysplasia / genetics*
  • Cineangiography / standards
  • Cineangiography / statistics & numerical data
  • Cohort Studies
  • Desmocollins / genetics
  • Desmoglein 2 / genetics
  • Diagnostic Techniques, Cardiovascular / standards*
  • Diagnostic Techniques, Cardiovascular / statistics & numerical data*
  • Electrocardiography, Ambulatory / standards
  • Electrocardiography, Ambulatory / statistics & numerical data
  • Exercise Test / standards
  • Exercise Test / statistics & numerical data
  • Family Health
  • Humans
  • Magnetic Resonance Imaging / standards
  • Magnetic Resonance Imaging / statistics & numerical data
  • Middle Aged
  • Mutation
  • Plakophilins / genetics
  • Sensitivity and Specificity
  • Young Adult

Substances

  • DSC2 protein, human
  • DSG2 protein, human
  • Desmocollins
  • Desmoglein 2
  • PKP2 protein, human
  • Plakophilins