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Design and rationale of a prospective, collaborative meta-analysis of all randomized controlled trials of angiotensin receptor antagonists in Marfan syndrome, based on individual patient data: A report from the Marfan Treatment Trialists' Collaboration.
Am Heart J. 2015 May; 169(5):605-12.AH

Abstract

RATIONALE

A number of randomized trials are underway, which will address the effects of angiotensin receptor blockers (ARBs) on aortic root enlargement and a range of other end points in patients with Marfan syndrome. If individual participant data from these trials were to be combined, a meta-analysis of the resulting data, totaling approximately 2,300 patients, would allow estimation across a number of trials of the treatment effects both of ARB therapy and of β-blockade. Such an analysis would also allow estimation of treatment effects in particular subgroups of patients on a range of end points of interest and would allow a more powerful estimate of the effects of these treatments on a composite end point of several clinical outcomes than would be available from any individual trial.

DESIGN

A prospective, collaborative meta-analysis based on individual patient data from all randomized trials in Marfan syndrome of (i) ARBs versus placebo (or open-label control) and (ii) ARBs versus β-blockers will be performed. A prospective study design, in which the principal hypotheses, trial eligibility criteria, analyses, and methods are specified in advance of the unblinding of the component trials, will help to limit bias owing to data-dependent emphasis on the results of particular trials. The use of individual patient data will allow for analysis of the effects of ARBs in particular patient subgroups and for time-to-event analysis for clinical outcomes. The meta-analysis protocol summarized in this report was written on behalf of the Marfan Treatment Trialists' Collaboration and finalized in late 2012, without foreknowledge of the results of any component trial, and will be made available online (http://www.ctsu.ox.ac.uk/research/meta-trials).

Links

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Authors+Show Affiliations

Pitcher A
Division of Cardiovascular Medicine, Radcliffe Department of Medicine, University of Oxford, Oxford, UK. Electronic address: alex.pitcher@cardiov.ox.ac.uk.
Emberson J
Clinical Trial Service Unit & Epidemiological Studies Unit, University of Oxford, Oxford, UK.
Lacro RV
Children's Hospital Boston and Harvard Medical School, Boston, MA.
Sleeper LA
New England Research Institutes, Watertown, MA.
Stylianou M
National Heart, Lung, and Blood Institute, NIH, Bethesda, MD.
Mahony L
University of Texas Southwestern Medical Center, Dallas, TX.
Pearson GD
National Heart, Lung, and Blood Institute, NIH, Bethesda, MD.
Groenink M
Academic Medical Center, University of Amsterdam, Amsterdam, The Netherlands.
Mulder BJ
Academic Medical Center, University of Amsterdam, Amsterdam, The Netherlands.
Zwinderman AH
Academic Medical Center, University of Amsterdam, Amsterdam, The Netherlands.
De Backer J
Center for Medical Genetics and Department of Cardiology, University Hospital Ghent, Ghent, Belgium.
De Paepe AM
Center for Medical Genetics, University Hospital Ghent, Ghent, Belgium.
Arbustini E
Centre for Inherited Cardiovascular Diseases, IRCCS Foundation, San Matteo Hospital, Pavia, Italy.
Erdem G
Clinical Trials and Evaluation Unit, Royal Brompton & Harefield NHS Trust, London, UK.
Jin XY
John Radcliffe Hospital, Oxford, UK.
Flather MD
Faculty of Medicine and Health Sciences, University of East Anglia, Norwich, UK.
Mullen MJ
UCL Institute of Cardiovascular Science, The Heart Hospital, London, UK.
Child AH
Department of Cardiac and Vascular Sciences, St George's Hospital, University of London, London, UK.
Forteza A
Hospital Universitario 12 de Octubre, Madrid, Spain.
Evangelista A
Marfan Sydrome Unit, Department of Cardiology, Hospital Vall d'Hebron, Barcelona, Spain.
Chiu HH
Department of Pediatrics and Adult Congenital Heart Center, Taipei Medical University Hospital, Taipei, Taiwan.
Wu MH
Department of Pediatrics and Adult Congenital Heart Center, National Taiwan University Hospital, Taipei, Taiwan.
Sandor G
Children's Heart Centre, British Columbia's Children's Hospital, Vancouver, British Columbia, Canada.
Bhatt AB
Massachusetts General Hospital, Boston, MA.
Creager MA
Brigham and Women's Hospital and Harvard Medical School, Boston, MA.
Devereux RB
Weill Cornell Medical College and New York-Presbyterian Hospital, New York, NY.
Loeys B
Center for Medical Genetics, Faculty of Medicine and Health Sciences, University of Antwerp and Antwerp University Hospital, Antwerp, Belgium.
Forfar JC
John Radcliffe Hospital, Oxford, UK.
Neubauer S
Oxford Centre for Clinical Magnetic Resonance Research, Division of Cardiovascular Medicine, Radcliffe Department of Medicine, University of Oxford, Oxford, UK.
Watkins H
Radcliffe Department of Medicine, University of Oxford, Oxford, UK.
Boileau C
Inserm LVTS U1148, Departement de Génétique, Hôpital Xavier Bichat-Claude Bernard, Paris, France.
Jondeau G
Centre National de Référence pour le syndrome de Marfan et apparentés, INSERM LVTS U1148, Service de Cardiologie, AP-HP Hôpital Bichat, Paris, France.
Dietz HC
McKusick-Nathans Institute of Genetic Medicine, Johns Hopkins University School of Medicine, and the Howard Hughes Medical Institute, Baltimore, MD.
Baigent C
Clinical Trial Service Unit & Epidemiological Studies Unit, University of Oxford, Oxford, UK.

MeSH

Angiotensin Receptor AntagonistsFemaleHumansMaleMarfan SyndromeMeta-Analysis as TopicProspective StudiesRandomized Controlled Trials as TopicResearch Design

Pub Type(s)

Journal Article
Research Support, Non-U.S. Gov't

Language

eng

PubMed ID

25965707

Citation

Pitcher, Alex, et al. "Design and Rationale of a Prospective, Collaborative Meta-analysis of All Randomized Controlled Trials of Angiotensin Receptor Antagonists in Marfan Syndrome, Based On Individual Patient Data: a Report From the Marfan Treatment Trialists' Collaboration." American Heart Journal, vol. 169, no. 5, 2015, pp. 605-12.
Pitcher A, Emberson J, Lacro RV, et al. Design and rationale of a prospective, collaborative meta-analysis of all randomized controlled trials of angiotensin receptor antagonists in Marfan syndrome, based on individual patient data: A report from the Marfan Treatment Trialists' Collaboration. Am Heart J. 2015;169(5):605-12.
Pitcher, A., Emberson, J., Lacro, R. V., Sleeper, L. A., Stylianou, M., Mahony, L., Pearson, G. D., Groenink, M., Mulder, B. J., Zwinderman, A. H., De Backer, J., De Paepe, A. M., Arbustini, E., Erdem, G., Jin, X. Y., Flather, M. D., Mullen, M. J., Child, A. H., Forteza, A., ... Baigent, C. (2015). Design and rationale of a prospective, collaborative meta-analysis of all randomized controlled trials of angiotensin receptor antagonists in Marfan syndrome, based on individual patient data: A report from the Marfan Treatment Trialists' Collaboration. American Heart Journal, 169(5), 605-12. https://doi.org/10.1016/j.ahj.2015.01.011
Pitcher A, et al. Design and Rationale of a Prospective, Collaborative Meta-analysis of All Randomized Controlled Trials of Angiotensin Receptor Antagonists in Marfan Syndrome, Based On Individual Patient Data: a Report From the Marfan Treatment Trialists' Collaboration. Am Heart J. 2015;169(5):605-12. PubMed PMID: 25965707.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Design and rationale of a prospective, collaborative meta-analysis of all randomized controlled trials of angiotensin receptor antagonists in Marfan syndrome, based on individual patient data: A report from the Marfan Treatment Trialists' Collaboration. AU - Pitcher,Alex, AU - Emberson,Jonathan, AU - Lacro,Ronald V, AU - Sleeper,Lynn A, AU - Stylianou,Mario, AU - Mahony,Lynn, AU - Pearson,Gail D, AU - Groenink,Maarten, AU - Mulder,Barbara J, AU - Zwinderman,Aeilko H, AU - De Backer,Julie, AU - De Paepe,Anne M, AU - Arbustini,Eloisa, AU - Erdem,Guliz, AU - Jin,Xu Yu, AU - Flather,Marcus D, AU - Mullen,Michael J, AU - Child,Anne H, AU - Forteza,Alberto, AU - Evangelista,Arturo, AU - Chiu,Hsin-Hui, AU - Wu,Mei-Hwan, AU - Sandor,George, AU - Bhatt,Ami B, AU - Creager,Mark A, AU - Devereux,Richard B, AU - Loeys,Bart, AU - Forfar,J Colin, AU - Neubauer,Stefan, AU - Watkins,Hugh, AU - Boileau,Catherine, AU - Jondeau,Guillaume, AU - Dietz,Harry C, AU - Baigent,Colin, Y1 - 2015/02/12/ PY - 2014/07/27/received PY - 2015/01/17/accepted PY - 2015/5/13/entrez PY - 2015/5/13/pubmed PY - 2015/7/17/medline SP - 605 EP - 12 JF - American heart journal JO - Am Heart J VL - 169 IS - 5 N2 - RATIONALE: A number of randomized trials are underway, which will address the effects of angiotensin receptor blockers (ARBs) on aortic root enlargement and a range of other end points in patients with Marfan syndrome. If individual participant data from these trials were to be combined, a meta-analysis of the resulting data, totaling approximately 2,300 patients, would allow estimation across a number of trials of the treatment effects both of ARB therapy and of β-blockade. Such an analysis would also allow estimation of treatment effects in particular subgroups of patients on a range of end points of interest and would allow a more powerful estimate of the effects of these treatments on a composite end point of several clinical outcomes than would be available from any individual trial. DESIGN: A prospective, collaborative meta-analysis based on individual patient data from all randomized trials in Marfan syndrome of (i) ARBs versus placebo (or open-label control) and (ii) ARBs versus β-blockers will be performed. A prospective study design, in which the principal hypotheses, trial eligibility criteria, analyses, and methods are specified in advance of the unblinding of the component trials, will help to limit bias owing to data-dependent emphasis on the results of particular trials. The use of individual patient data will allow for analysis of the effects of ARBs in particular patient subgroups and for time-to-event analysis for clinical outcomes. The meta-analysis protocol summarized in this report was written on behalf of the Marfan Treatment Trialists' Collaboration and finalized in late 2012, without foreknowledge of the results of any component trial, and will be made available online (http://www.ctsu.ox.ac.uk/research/meta-trials). SN - 1097-6744 UR - https://www.unboundmedicine.com/medline/citation/25965707/Design_and_rationale_of_a_prospective_collaborative_meta_analysis_of_all_randomized_controlled_trials_of_angiotensin_receptor_antagonists_in_Marfan_syndrome_based_on_individual_patient_data:_A_report_from_the_Marfan_Treatment_Trialists'_Collaboration_ DB - PRIME DP - Unbound Medicine ER -