Yield and Pitfalls of Ajmaline Testing in the Evaluation of Unexplained Cardiac Arrest and Sudden Unexplained Death: Single-Center Experience With 482 Families

JACC Clin Electrophysiol. 2017 Dec 11;3(12):1400-1408. doi: 10.1016/j.jacep.2017.04.005. Epub 2017 Jun 28.

Abstract

Objectives: This study evaluated the yield of ajmaline testing and assessed the occurrence of confounding responses in a large cohort of families with unexplained cardiac arrest (UCA) or sudden unexplained death (SUD).

Background: Ajmaline testing to diagnose Brugada syndrome (BrS) is routinely used in the evaluation of SUD and UCA, but its yield, limitations, and appropriate dosing have not been studied in a large cohort.

Methods: We assessed ajmaline test response and genetic testing results in 637 individuals from 482 families who underwent ajmaline testing for SUD or UCA.

Results: Overall, 89 individuals (14%) from 88 families (18%) had a positive ajmaline test result. SCN5A mutations were identified in 9 of 86 ajmaline-positive cases (10%). SCN5A mutation carriers had positive test results at significantly lower ajmaline doses than noncarriers (0.75 [range: 0.64 to 0.98] mg/kg vs. 1.03 [range: 0.95 to 1.14] mg/kg, respectively; p < 0.01). In 7 of 88 families (8%), it was concluded that the positive ajmaline response was a confounder, either in the presence of an alternative genetic diagnosis accounting for UCA/SUD (5 cases) or noncosegregation of positive ajmaline response and arrhythmia (2 cases). The rate of confounding responses was significantly higher in positive ajmaline responses obtained at >1 mg/kg than in those obtained at ≤1 mg/kg (7 of 48 vs. 0 of 41 individuals; Fisher's exact test: p = 0.014).

Conclusions: In line with previous, smaller studies, a positive ajmaline response was observed in a large proportion of UCA/SUD families. Importantly, our data emphasize the potential for confounding possibly false-positive ajmaline responses in this population, particularly at high doses, which could possibly lead to a misdiagnosis. Clinicians should consider all alternative causes in UCA/SUD and avoid ajmaline doses >1 mg/kg.

Keywords: Brugada syndrome; sodium channel blocker; sudden cardiac death; unexplained cardiac arrest.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Administration, Intravenous
  • Adult
  • Ajmaline / administration & dosage*
  • Brugada Syndrome / diagnosis*
  • Brugada Syndrome / epidemiology
  • Brugada Syndrome / genetics
  • Death, Sudden, Cardiac / epidemiology
  • Death, Sudden, Cardiac / etiology*
  • Diagnostic Errors / prevention & control
  • Female
  • Genetic Testing / methods
  • Heart Arrest / epidemiology
  • Heart Arrest / etiology*
  • Heterozygote
  • Humans
  • Male
  • Middle Aged
  • Mutation
  • NAV1.5 Voltage-Gated Sodium Channel / genetics
  • Pedigree
  • Phenotype
  • Ventricular Fibrillation / diagnosis
  • Ventricular Fibrillation / epidemiology
  • Ventricular Fibrillation / physiopathology
  • Voltage-Gated Sodium Channel Blockers

Substances

  • NAV1.5 Voltage-Gated Sodium Channel
  • SCN5A protein, human
  • Voltage-Gated Sodium Channel Blockers
  • Ajmaline