Valente Anne Marie, Gauvreau Kimberlee, Assenza Gabriele Egidy, Babu-Narayan Sonya V, Evans Sarah P, Gatzoulis Michael, Groenink Maarten, Inuzuka Ryo, Kilner Philip J, Koyak Zeliha, Landzberg Michael J, Mulder Barbara, Powell Andrew J, Wald Rachel, Geva Tal
Department of Cardiology, Boston Children's Hospital and Department of Pediatrics, Harvard Medical School, Boston, MA 02115, USA.
Pediatr Cardiol. 2013 Jan;34(1):95-104. doi: 10.1007/s00246-012-0394-5. Epub 2012 Jun 5.
Although early survival after tetralogy of Fallot (TOF) repair in the modern era is excellent, studies on late outcomes have shown increasing rates of mortality and morbidity. Despite multiple publications on factors associated with late complications, risk factors for major outcomes (death and sustained ventricular tachycardia [VT]) remain poorly defined. Consequently, the International Multicenter TOF Registry (INDICATOR) was established. This article describes the development, structure, and goals of this registry and characterizes the initial cohort derived from four large congenital heart centers in the United States, Canada, and Europe. A data coordinating center with a core cardiac magnetic resonance (CMR) laboratory and statistical core was established. Subjects with repaired TOF who had CMR imaging performed between 1997 and 2010 and ≥ 1 year follow-up were included. Clinical end points were death and sustained VT. Demographic, electrophysiologic, exercise, and outcome data were collected. A total of 873 subjects fulfilled inclusion criteria (median age at repair 2.9 years and at CMR imaging 22.8 years). Of these, 9 % had QRS duration >180 ms on electrocardiogram (ECG). On CMR imaging, 38 % had severe right-ventricular (RV) dilatation (≥ 160 mL/m(2)), and 6 % had severe RV dysfunction (ejection fraction < 35 %). Of the 551 subjects with exercise testing available, 28 % had severely decreased exercise capacity with <50 % predicted peak oxygen consumption. The INDICATOR cohort allows robust statistical analysis to evaluate major clinical outcomes in patients with repaired TOF. Continued follow-up and further expansion of the registry may provide new insights into innovative therapeutic strategies to improve late outcomes.
尽管现代法洛四联症(TOF)修复术后的早期生存率很高,但关于晚期结局的研究表明,死亡率和发病率在不断上升。尽管有多项关于晚期并发症相关因素的出版物,但主要结局(死亡和持续性室性心动过速[VT])的危险因素仍不明确。因此,建立了国际多中心TOF注册研究(INDICATOR)。本文描述了该注册研究的发展、结构和目标,并对来自美国、加拿大和欧洲四个大型先天性心脏病中心的初始队列进行了特征描述。建立了一个数据协调中心,该中心设有一个核心心脏磁共振(CMR)实验室和统计核心。纳入1997年至2010年间接受CMR成像且随访≥1年的TOF修复患者。临床终点为死亡和持续性VT。收集了人口统计学、电生理学、运动和结局数据。共有873名受试者符合纳入标准(修复时的中位年龄为2.9岁,CMR成像时为22.8岁)。其中,9%的受试者心电图(ECG)上QRS时限>180 ms。在CMR成像中,38%的受试者右心室(RV)严重扩张(≥160 mL/m²),6%的受试者右心室功能严重障碍(射血分数<35%)。在551名可进行运动测试的受试者中,28%的受试者运动能力严重下降,预测的峰值耗氧量<50%。INDICATOR队列允许进行有力的统计分析,以评估TOF修复患者的主要临床结局。对该注册研究的持续随访和进一步扩展可能会为改善晚期结局的创新治疗策略提供新的见解。