TY - JOUR
T1 - Repeatability of ventricular arrhythmia characteristics on the exercise-stress test in RYR2-mediated catecholaminergic polymorphic ventricular tachycardia
AU - Peltenburg, Puck J.
AU - Pultoo, Sanjeev N. J.
AU - Tobert, Kathryn E.
AU - Bos, J. Martijn
AU - Lieve, Krystien V. V.
AU - Tanck, Michael
AU - Clur, Sally-Ann B.
AU - Blom, Nico A.
AU - Ackerman, Michael J.
AU - Wilde, Arthur A. M.
AU - van der Werf, Christian
N1 - Publisher Copyright: © The Author(s) 2022. Published by Oxford University Press on behalf of the European Society of Cardiology. Funding Information: This work was supported by ZonMW Priority Medicines for Rare Diseases and Orphan Drugs (grant 113304045 to C.v.d.W.), the Mayo Clinic Windland Smith Rice Comprehensive Sudden Cardiac Death Program (to M.J.A.), the Netherlands CardioVascular Research Initiative: the Dutch Heart Foundation, the Nederlandse Federatie van Universitair Medische Centra, the Netherlands Organisation for Health Research and Development and the Royal Netherlands Academy of Sciences (CVON 2012-10 PREDICT to A.A.M.W.), E-Rare Joint Transnational Call for Proposals 2015 'Improving Diagnosis and Treatment of Catecholaminergic Polymorphic Ventricular Tachycardia: Integrating Clinical and Basic Science' (to A.A.M.W.), and the AEPC junior members research grant 2019 (to P.J.P.). Publisher Copyright: © 2022 The Author(s).
PY - 2023/2/1
Y1 - 2023/2/1
N2 - AIMS: In catecholaminergic polymorphic ventricular tachycardia (CPVT), the exercise-stress test (EST) is the cornerstone for the diagnosis, risk stratification, and assessment of therapeutic efficacy, but its repeatability is unknown. We aimed to test the repeatability of ventricular arrhythmia characteristics on the EST in patients with CPVT. METHODS AND RESULTS: EST-pairs (ESTs performed within 18 months between 2005 and 2021, on the same protocol, and without or on the exact same treatment) of patients with RYR2-mediated CPVT from two specialized centres were included. The primary endpoint was the repeatability of the maximum ventricular arrhythmia score [VAS: 0 for the absence of premature ventricular contractions (PVCs); 1 for isolated PVCs; 2 for bigeminal PVCs; 3 for couplets; and 4 for non-sustained ventricular tachycardia]. Secondary outcomes were the repeatability of the heart rate at the first PVC and the ΔVAS (the absolute difference in VAS between the EST-pairs). A total of 104 patients with 349 EST-pairs were included. The median duration between ESTs was 343 (interquartile range, 189-378) days. Sixty (17.2%) EST-pairs were off therapy. The repeatability of the VAS was moderate {Krippendorf α, 0.56 [95% confidence interval (CI), 0.48-0.64]}, and the repeatability of the heart rate at the first PVC was substantial [intra-class correlation coefficient, 0.78 (95% CI, 0.71-0.84)]. The use of medication was associated with a higher odds for a ΔVAS > 1 (odds ratio = 3.52; 95% CI, 2.46-4.57; P = 0.020). CONCLUSION: The repeatability of ventricular arrhythmia characteristics was moderate to substantial. This underlines the need for multiple ESTs in CPVT patients and CPVT suspicious patients and it provides the framework for assessing the therapeutic efficacy of novel CPVT therapies.
AB - AIMS: In catecholaminergic polymorphic ventricular tachycardia (CPVT), the exercise-stress test (EST) is the cornerstone for the diagnosis, risk stratification, and assessment of therapeutic efficacy, but its repeatability is unknown. We aimed to test the repeatability of ventricular arrhythmia characteristics on the EST in patients with CPVT. METHODS AND RESULTS: EST-pairs (ESTs performed within 18 months between 2005 and 2021, on the same protocol, and without or on the exact same treatment) of patients with RYR2-mediated CPVT from two specialized centres were included. The primary endpoint was the repeatability of the maximum ventricular arrhythmia score [VAS: 0 for the absence of premature ventricular contractions (PVCs); 1 for isolated PVCs; 2 for bigeminal PVCs; 3 for couplets; and 4 for non-sustained ventricular tachycardia]. Secondary outcomes were the repeatability of the heart rate at the first PVC and the ΔVAS (the absolute difference in VAS between the EST-pairs). A total of 104 patients with 349 EST-pairs were included. The median duration between ESTs was 343 (interquartile range, 189-378) days. Sixty (17.2%) EST-pairs were off therapy. The repeatability of the VAS was moderate {Krippendorf α, 0.56 [95% confidence interval (CI), 0.48-0.64]}, and the repeatability of the heart rate at the first PVC was substantial [intra-class correlation coefficient, 0.78 (95% CI, 0.71-0.84)]. The use of medication was associated with a higher odds for a ΔVAS > 1 (odds ratio = 3.52; 95% CI, 2.46-4.57; P = 0.020). CONCLUSION: The repeatability of ventricular arrhythmia characteristics was moderate to substantial. This underlines the need for multiple ESTs in CPVT patients and CPVT suspicious patients and it provides the framework for assessing the therapeutic efficacy of novel CPVT therapies.
KW - Catecholaminergic polymorphic ventricular tachycardia
KW - Diagnostic test
KW - Exercise-stress test
KW - RYR2
KW - Ventricular arrhythmia
UR - http://www.scopus.com/inward/record.url?scp=85148307479&partnerID=8YFLogxK
U2 - https://doi.org/10.1093/europace/euac177
DO - https://doi.org/10.1093/europace/euac177
M3 - Article
C2 - 36369981
SN - 1099-5129
VL - 25
SP - 619
EP - 626
JO - Europace : European pacing, arrhythmias, and cardiac electrophysiology
JF - Europace : European pacing, arrhythmias, and cardiac electrophysiology
IS - 2
ER -