Skip to main content
Top
Published in: Trials 1/2011

Open Access 01-12-2011 | Research

Designs for clinical trials with time-to-event outcomes based on stopping guidelines for lack of benefit

Authors: Patrick Royston, Friederike M-S Barthel, Mahesh KB Parmar, Babak Choodari-Oskooei, Valerie Isham

Published in: Trials | Issue 1/2011

Login to get access

Abstract

background

The pace of novel medical treatments and approaches to therapy has accelerated in recent years. Unfortunately, many potential therapeutic advances do not fulfil their promise when subjected to randomized controlled trials. It is therefore highly desirable to speed up the process of evaluating new treatment options, particularly in phase II and phase III trials. To help realize such an aim, in 2003, Royston and colleagues proposed a class of multi-arm, two-stage trial designs intended to eliminate poorly performing contenders at a first stage (point in time). Only treatments showing a predefined degree of advantage against a control treatment were allowed through to a second stage. Arms that survived the first-stage comparison on an intermediate outcome measure entered a second stage of patient accrual, culminating in comparisons against control on the definitive outcome measure. The intermediate outcome is typically on the causal pathway to the definitive outcome (i.e. the features that cause an intermediate event also tend to cause a definitive event), an example in cancer being progression-free and overall survival. Although the 2003 paper alluded to multi-arm trials, most of the essential design features concerned only two-arm trials. Here, we extend the two-arm designs to allow an arbitrary number of stages, thereby increasing flexibility by building in several 'looks' at the accumulating data. Such trials can terminate at any of the intermediate stages or the final stage.

Methods

We describe the trial design and the mathematics required to obtain the timing of the 'looks' and the overall significance level and power of the design. We support our results by extensive simulation studies. As an example, we discuss the design of the STAMPEDE trial in prostate cancer.

Results

The mathematical results on significance level and power are confirmed by the computer simulations. Our approach compares favourably with methodology based on beta spending functions and on monitoring only a primary outcome measure for lack of benefit of the new treatment.

Conclusions

The new designs are practical and are supported by theory. They hold considerable promise for speeding up the evaluation of new treatments in phase II and III trials.
Literature
1.
go back to reference US Food and Drug Administration: Innovation or Stagnation: Challenge and Opportunity on the Critical Path to New Medical Products. US Dept of Health and Human Services. 2004 US Food and Drug Administration: Innovation or Stagnation: Challenge and Opportunity on the Critical Path to New Medical Products. US Dept of Health and Human Services. 2004
2.
go back to reference Proschan MA, Lan KKG, Wittes J: Statistical Monitoring of Clinical Trials - A Unified Approach. 2006, New York: Springer Proschan MA, Lan KKG, Wittes J: Statistical Monitoring of Clinical Trials - A Unified Approach. 2006, New York: Springer
3.
go back to reference Armitage P, McPherson CK, Rowe BC: Repeated significance tests on accumulating data. Journal of the Royal Statistical Society, Series A. 1969, 132: 235-244. 10.2307/2343787.CrossRef Armitage P, McPherson CK, Rowe BC: Repeated significance tests on accumulating data. Journal of the Royal Statistical Society, Series A. 1969, 132: 235-244. 10.2307/2343787.CrossRef
4.
go back to reference Lan K, DeMets D: Discrete sequential boundaries for clinical trials. Biometrika. 1983, 70: 659-663. 10.2307/2336502.CrossRef Lan K, DeMets D: Discrete sequential boundaries for clinical trials. Biometrika. 1983, 70: 659-663. 10.2307/2336502.CrossRef
5.
go back to reference O'Brien PC, Fleming TR: A multiple testing procedure for clinical trials. Biometrics. 1979, 35: 549-556.CrossRefPubMed O'Brien PC, Fleming TR: A multiple testing procedure for clinical trials. Biometrics. 1979, 35: 549-556.CrossRefPubMed
6.
go back to reference Pampallona S, Tsiatis A, Kim KM: Interim monitoring of group sequential trials using spending functions for the type I and II error probabilities. Drug Information Journal. 2001, 35: 1113-1121.CrossRef Pampallona S, Tsiatis A, Kim KM: Interim monitoring of group sequential trials using spending functions for the type I and II error probabilities. Drug Information Journal. 2001, 35: 1113-1121.CrossRef
7.
go back to reference Royston P, Parmar MKB, Qian W: Novel designs for multi-arm clinical trials with survival outcomes, with an application in ovarian cancer. Statistics in Medicine. 2003, 22: 2239-2256. 10.1002/sim.1430.CrossRefPubMed Royston P, Parmar MKB, Qian W: Novel designs for multi-arm clinical trials with survival outcomes, with an application in ovarian cancer. Statistics in Medicine. 2003, 22: 2239-2256. 10.1002/sim.1430.CrossRefPubMed
8.
go back to reference Bookman MA, Brady MF, McGuire WP, Harper PG, Alberts DS, Friedlander M, Colombo N, Fowler JM, Argenta PA, Geest KD, Mutch DG, Burger RA, Swart AM, Trimble EL, Accario-Winslow C, Roth LM: Evaluation of New Platinum-Based Treatment Regimens in Advanced-Stage Ovarian Cancer: A Phase III Trial of the Gynecologic Cancer InterGroup. Journal of Clinical Oncology. 2009, 27: 1419-1425. 10.1200/JCO.2008.19.1684.CrossRefPubMedPubMedCentral Bookman MA, Brady MF, McGuire WP, Harper PG, Alberts DS, Friedlander M, Colombo N, Fowler JM, Argenta PA, Geest KD, Mutch DG, Burger RA, Swart AM, Trimble EL, Accario-Winslow C, Roth LM: Evaluation of New Platinum-Based Treatment Regimens in Advanced-Stage Ovarian Cancer: A Phase III Trial of the Gynecologic Cancer InterGroup. Journal of Clinical Oncology. 2009, 27: 1419-1425. 10.1200/JCO.2008.19.1684.CrossRefPubMedPubMedCentral
9.
go back to reference James ND, Sydes MR, Clarke NW, Mason MD, Dearnaley DP, Anderson J, Popert RJ, Sanders K, Morgan RC, Stansfeld J, Dwyer J, Masters J, Parmar MKB: STAMPEDE: Systemic Therapy for Advancing or Metastatic Prostate Cancer - A Multi-Arm Multi-Stage Randomised Controlled Trial. Clinical Oncology. 2008, 20: 577-581. 10.1016/j.clon.2008.07.002.CrossRefPubMed James ND, Sydes MR, Clarke NW, Mason MD, Dearnaley DP, Anderson J, Popert RJ, Sanders K, Morgan RC, Stansfeld J, Dwyer J, Masters J, Parmar MKB: STAMPEDE: Systemic Therapy for Advancing or Metastatic Prostate Cancer - A Multi-Arm Multi-Stage Randomised Controlled Trial. Clinical Oncology. 2008, 20: 577-581. 10.1016/j.clon.2008.07.002.CrossRefPubMed
10.
go back to reference Tsiatis AA: The asymptotic joint distribution of the efficient scores test for the propor- tional hazards model calculated over time. Biometrika. 1981, 68: 311-315. 10.1093/biomet/68.1.311.CrossRef Tsiatis AA: The asymptotic joint distribution of the efficient scores test for the propor- tional hazards model calculated over time. Biometrika. 1981, 68: 311-315. 10.1093/biomet/68.1.311.CrossRef
11.
go back to reference Betensky R: Construction of a continuous stopping boundary from an alpha spending function. Biometrics. 1998, 54: 1061-1071. 10.2307/2533857.CrossRefPubMed Betensky R: Construction of a continuous stopping boundary from an alpha spending function. Biometrics. 1998, 54: 1061-1071. 10.2307/2533857.CrossRefPubMed
12.
go back to reference Freidlin B, Korn EL, Gray R: A general inefficacy interim monitoring rule for randomized clinical trials. Clinical Trials. 2010, 7: 197-208. 10.1177/1740774510369019.CrossRefPubMed Freidlin B, Korn EL, Gray R: A general inefficacy interim monitoring rule for randomized clinical trials. Clinical Trials. 2010, 7: 197-208. 10.1177/1740774510369019.CrossRefPubMed
13.
go back to reference Royston P, Wright EM: A method for estimating age-specific reference intervals ("normal ranges") based on fractional polynomials and exponential transformation. Journal of the Royal Statistical Society, Series A. 1998, 161: 79-101.CrossRef Royston P, Wright EM: A method for estimating age-specific reference intervals ("normal ranges") based on fractional polynomials and exponential transformation. Journal of the Royal Statistical Society, Series A. 1998, 161: 79-101.CrossRef
14.
go back to reference Prentice RL: Surrogate endpoints in clinical trials: definition and operational criteria. Statistics in Medicine. 1989, 8: 431-440. 10.1002/sim.4780080407.CrossRefPubMed Prentice RL: Surrogate endpoints in clinical trials: definition and operational criteria. Statistics in Medicine. 1989, 8: 431-440. 10.1002/sim.4780080407.CrossRefPubMed
15.
go back to reference Barthel FMS, Babiker A, Royston P, Parmar MKB: Evaluation of sample size and power for multi-arm survival trials allowing for non-uniform accrual, non-proportional hazards, loss to follow-up and cross-over. Statistics in Medicine. 2006, 25: 2521-2542. 10.1002/sim.2517.CrossRefPubMed Barthel FMS, Babiker A, Royston P, Parmar MKB: Evaluation of sample size and power for multi-arm survival trials allowing for non-uniform accrual, non-proportional hazards, loss to follow-up and cross-over. Statistics in Medicine. 2006, 25: 2521-2542. 10.1002/sim.2517.CrossRefPubMed
16.
go back to reference Royston P, Parmar MKB: Flexible Parametric Proportional-Hazards and Proportional-Odds Models for Censored Survival Data, with Application to Prognostic Modelling and Estimation of Treatment Effects. Statistics in Medicine. 2002, 21: 2175-2197. 10.1002/sim.1203.CrossRefPubMed Royston P, Parmar MKB: Flexible Parametric Proportional-Hazards and Proportional-Odds Models for Censored Survival Data, with Application to Prognostic Modelling and Estimation of Treatment Effects. Statistics in Medicine. 2002, 21: 2175-2197. 10.1002/sim.1203.CrossRefPubMed
17.
go back to reference Royston P: Flexible parametric alternatives to the Cox model, and more. Stata Journal. 2001, 1: 1-28. Royston P: Flexible parametric alternatives to the Cox model, and more. Stata Journal. 2001, 1: 1-28.
18.
go back to reference Lambert PC, Royston P: Further development of flexible parametric models for survival analysis. Stata Journal. 2009, 9: 265-290. Lambert PC, Royston P: Further development of flexible parametric models for survival analysis. Stata Journal. 2009, 9: 265-290.
19.
go back to reference Posch M, Bauer P, Brannath W: Issues in Designing Flexible Trials. Statistics in Medicine. 2003, 22: 953-969. 10.1002/sim.1455.CrossRefPubMed Posch M, Bauer P, Brannath W: Issues in Designing Flexible Trials. Statistics in Medicine. 2003, 22: 953-969. 10.1002/sim.1455.CrossRefPubMed
20.
go back to reference Cox DR, Isham V: Point Processes. 1980, London: Chapman and Hall Cox DR, Isham V: Point Processes. 1980, London: Chapman and Hall
Metadata
Title
Designs for clinical trials with time-to-event outcomes based on stopping guidelines for lack of benefit
Authors
Patrick Royston
Friederike M-S Barthel
Mahesh KB Parmar
Babak Choodari-Oskooei
Valerie Isham
Publication date
01-12-2011
Publisher
BioMed Central
Published in
Trials / Issue 1/2011
Electronic ISSN: 1745-6215
DOI
https://doi.org/10.1186/1745-6215-12-81

Other articles of this Issue 1/2011

Trials 1/2011 Go to the issue