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Prospective risk stratification of sudden cardiac death in Marfan's syndrome

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Prospective risk stratification of sudden cardiac death in Marfan's syndrome. / Hoffmann, Boris A; Rybczynski, Meike; Rostock, Thomas; Servatius, Helge; Drewitz, Imke; Steven, Daniel; Aydin, Ali; Sheikhzadeh, Sara; Darko, Vivien; von Kodolitsch, Yskert; Willems, Stephan.

In: INT J CARDIOL, Vol. 167, No. 6, 10.09.2013, p. 2539-2545.

Research output: SCORING: Contribution to journal › SCORING: Journal article › Research › peer-review

Harvard

Hoffmann, BA, Rybczynski, M, Rostock, T, Servatius, H, Drewitz, I, Steven, D, Aydin, A, Sheikhzadeh, S, Darko, V, von Kodolitsch, Y & Willems, S 2013, 'Prospective risk stratification of sudden cardiac death in Marfan's syndrome', INT J CARDIOL, vol. 167, no. 6, pp. 2539-2545. https://doi.org/10.1016/j.ijcard.2012.06.036

APA

Hoffmann, B. A., Rybczynski, M., Rostock, T., Servatius, H., Drewitz, I., Steven, D., Aydin, A., Sheikhzadeh, S., Darko, V., von Kodolitsch, Y., & Willems, S. (2013). Prospective risk stratification of sudden cardiac death in Marfan's syndrome. INT J CARDIOL, 167(6), 2539-2545. https://doi.org/10.1016/j.ijcard.2012.06.036

Vancouver

Hoffmann BA, Rybczynski M, Rostock T, Servatius H, Drewitz I, Steven D et al. Prospective risk stratification of sudden cardiac death in Marfan's syndrome. INT J CARDIOL. 2013 Sep 10;167(6):2539-2545. https://doi.org/10.1016/j.ijcard.2012.06.036

Bibtex

@article{fa04e098d2054401b0f7a97fabe8b583,
title = "Prospective risk stratification of sudden cardiac death in Marfan's syndrome",
abstract = "BACKGROUND: Marfan syndrome (MFS) is a variable, autosomal-dominant disorder of the connective tissue. In MFS serious ventricular arrhythmias and sudden cardiac death (SCD) can occur. The aim of this prospective study was to reveal underlying risk factors and to prospectively investigate the association between MFS and SCD in a long-term follow-up.METHODS: 77 patients with MFS were included. At baseline serum N-terminal pro-brain natriuretic peptide (NT-proBNP), transthoracic echocardiogram, 12-lead resting ECG, signal-averaged ECG (SAECG) and a 24-h Holter ECG with time- and frequency domain analyses were performed. The primary composite endpoint was defined as SCD, ventricular tachycardia (VT), ventricular fibrillation (VF) or arrhythmogenic syncope.RESULTS: The median follow-up (FU) time was 868 days. Among all risk stratification parameters, NT-proBNP remained the exclusive predictor (hazard ratio [HR]: 2.34, 95% confidence interval [CI]: 1.1 to 4.62, p=0.01) for the composite endpoint. With an optimal cut-off point at 214.3 pg/ml NT-proBNP predicted the composite primary endpoint accurately (AUC 0.936, p=0.00046, sensitivity 100%, specificity 79.0%). During FU, seven patients of Group 2 (NT-proBNP ≥ 214.3 pg/ml) reached the composite endpoint and 2 of these patients died due to SCD. In five patients, sustained VT was documented. All patients with a NT-proBNP<214.3 pg/ml (Group 1) experienced no events. Group 2 patients had a significantly higher risk of experiencing the composite endpoint (logrank-test, p<0.001).CONCLUSIONS: In contrast to non-invasive electrocardiographic parameter, NT-proBNP independently predicts adverse arrhythmogenic events in patients with MFS.",
keywords = "Adult, Death, Sudden, Cardiac/epidemiology, Female, Follow-Up Studies, Humans, Male, Marfan Syndrome/diagnostic imaging, Middle Aged, Prospective Studies, Risk Assessment, Ultrasonography, Young Adult",
author = "Hoffmann, {Boris A} and Meike Rybczynski and Thomas Rostock and Helge Servatius and Imke Drewitz and Daniel Steven and Ali Aydin and Sara Sheikhzadeh and Vivien Darko and {von Kodolitsch}, Yskert and Stephan Willems",
note = "Copyright {\textcopyright} 2012 Elsevier Ireland Ltd. All rights reserved.",
year = "2013",
month = sep,
day = "10",
doi = "10.1016/j.ijcard.2012.06.036",
language = "English",
volume = "167",
pages = "2539--2545",
journal = "INT J CARDIOL",
issn = "0167-5273",
publisher = "Elsevier Ireland Ltd",
number = "6",

}

RIS

TY - JOUR

T1 - Prospective risk stratification of sudden cardiac death in Marfan's syndrome

AU - Hoffmann, Boris A

AU - Rybczynski, Meike

AU - Rostock, Thomas

AU - Servatius, Helge

AU - Drewitz, Imke

AU - Steven, Daniel

AU - Aydin, Ali

AU - Sheikhzadeh, Sara

AU - Darko, Vivien

AU - von Kodolitsch, Yskert

AU - Willems, Stephan

N1 - Copyright © 2012 Elsevier Ireland Ltd. All rights reserved.

PY - 2013/9/10

Y1 - 2013/9/10

N2 - BACKGROUND: Marfan syndrome (MFS) is a variable, autosomal-dominant disorder of the connective tissue. In MFS serious ventricular arrhythmias and sudden cardiac death (SCD) can occur. The aim of this prospective study was to reveal underlying risk factors and to prospectively investigate the association between MFS and SCD in a long-term follow-up.METHODS: 77 patients with MFS were included. At baseline serum N-terminal pro-brain natriuretic peptide (NT-proBNP), transthoracic echocardiogram, 12-lead resting ECG, signal-averaged ECG (SAECG) and a 24-h Holter ECG with time- and frequency domain analyses were performed. The primary composite endpoint was defined as SCD, ventricular tachycardia (VT), ventricular fibrillation (VF) or arrhythmogenic syncope.RESULTS: The median follow-up (FU) time was 868 days. Among all risk stratification parameters, NT-proBNP remained the exclusive predictor (hazard ratio [HR]: 2.34, 95% confidence interval [CI]: 1.1 to 4.62, p=0.01) for the composite endpoint. With an optimal cut-off point at 214.3 pg/ml NT-proBNP predicted the composite primary endpoint accurately (AUC 0.936, p=0.00046, sensitivity 100%, specificity 79.0%). During FU, seven patients of Group 2 (NT-proBNP ≥ 214.3 pg/ml) reached the composite endpoint and 2 of these patients died due to SCD. In five patients, sustained VT was documented. All patients with a NT-proBNP<214.3 pg/ml (Group 1) experienced no events. Group 2 patients had a significantly higher risk of experiencing the composite endpoint (logrank-test, p<0.001).CONCLUSIONS: In contrast to non-invasive electrocardiographic parameter, NT-proBNP independently predicts adverse arrhythmogenic events in patients with MFS.

AB - BACKGROUND: Marfan syndrome (MFS) is a variable, autosomal-dominant disorder of the connective tissue. In MFS serious ventricular arrhythmias and sudden cardiac death (SCD) can occur. The aim of this prospective study was to reveal underlying risk factors and to prospectively investigate the association between MFS and SCD in a long-term follow-up.METHODS: 77 patients with MFS were included. At baseline serum N-terminal pro-brain natriuretic peptide (NT-proBNP), transthoracic echocardiogram, 12-lead resting ECG, signal-averaged ECG (SAECG) and a 24-h Holter ECG with time- and frequency domain analyses were performed. The primary composite endpoint was defined as SCD, ventricular tachycardia (VT), ventricular fibrillation (VF) or arrhythmogenic syncope.RESULTS: The median follow-up (FU) time was 868 days. Among all risk stratification parameters, NT-proBNP remained the exclusive predictor (hazard ratio [HR]: 2.34, 95% confidence interval [CI]: 1.1 to 4.62, p=0.01) for the composite endpoint. With an optimal cut-off point at 214.3 pg/ml NT-proBNP predicted the composite primary endpoint accurately (AUC 0.936, p=0.00046, sensitivity 100%, specificity 79.0%). During FU, seven patients of Group 2 (NT-proBNP ≥ 214.3 pg/ml) reached the composite endpoint and 2 of these patients died due to SCD. In five patients, sustained VT was documented. All patients with a NT-proBNP<214.3 pg/ml (Group 1) experienced no events. Group 2 patients had a significantly higher risk of experiencing the composite endpoint (logrank-test, p<0.001).CONCLUSIONS: In contrast to non-invasive electrocardiographic parameter, NT-proBNP independently predicts adverse arrhythmogenic events in patients with MFS.

KW - Adult

KW - Death, Sudden, Cardiac/epidemiology

KW - Female

KW - Follow-Up Studies

KW - Humans

KW - Male

KW - Marfan Syndrome/diagnostic imaging

KW - Middle Aged

KW - Prospective Studies

KW - Risk Assessment

KW - Ultrasonography

KW - Young Adult

U2 - 10.1016/j.ijcard.2012.06.036

DO - 10.1016/j.ijcard.2012.06.036

M3 - SCORING: Journal article

C2 - 22738784

VL - 167

SP - 2539

EP - 2545

JO - INT J CARDIOL

JF - INT J CARDIOL

SN - 0167-5273

IS - 6

ER -