Evidence map›Paper›PMID 31761997›Full record

ArticlePharmacoEconomics2020

How Uncertain is the Survival Extrapolation? A Study of the Impact of Different Parametric Survival Models on Extrapolated Uncertainty About Hazard Functions, Lifetime Mean Survival and Cost Effectiveness.

Ben Kearns, John Stevens, Shijie Ren, Alan Brennan

Abstract read
In one paragraph

Article in PharmacoEconomics, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 25 papers, 4 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
25citing papers in PubMed, 4 pooled it
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

25 citing papers in PubMed, 4 syntheses or guidelines pooled it.

  1. Pooled it
  2. Pooled it
  3. Pooled it
  4. A Systematic Review of Methods to Incorporate External Evidence into Trial-Based Survival Extrapolations for Health Technology Assessment.Medical decision making : an international journal of the Society for Medical Decision Making · 2023
    Pooled it
  5. Article
  6. Article
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  8. Impact of Limited Sample Size and Follow-up on Partitioned Survival and Multistate Modeling-Based Health Economic Models: A Simulation Study.Medical decision making : an international journal of the Society for Medical Decision Making · 2025
    Article
  9. Article
  10. Article
  11. Article
  12. Article
  13. Survival Extrapolation Incorporating General Population Mortality Using Excess Hazard and Cure Models: A Tutorial.Medical decision making : an international journal of the Society for Medical Decision Making · 2023
    Review
  14. Blended Survival Curves: A New Approach to Extrapolation for Time-to-Event Outcomes from Clinical Trials in Health Technology Assessment.Medical decision making : an international journal of the Society for Medical Decision Making · 2023
    Article
  15. Article
  16. Article
  17. Dynamic and Flexible Survival Models for Extrapolation of Relative Survival: A Case Study and Simulation Study.Medical decision making : an international journal of the Society for Medical Decision Making · 2022
    Article
  18. Informed Bayesian survival analysis.BMC medical research methodology · 2022
    Article
  19. Article
  20. Article
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

4 authors.

Ben KearnsSchool of Health and Related Research (ScHARR), University of Sheffield, Regent Court, 30 Regent Street, Sheffield, S1 4DA, UK. b.kearns@sheffield.ac.uk.ORCID 0000-0001-7730-668X
John StevensSchool of Health and Related Research (ScHARR), University of Sheffield, Regent Court, 30 Regent Street, Sheffield, S1 4DA, UK.ORCID 0000-0002-9867-7209
Shijie RenSchool of Health and Related Research (ScHARR), University of Sheffield, Regent Court, 30 Regent Street, Sheffield, S1 4DA, UK.ORCID 0000-0003-3568-7124
Alan BrennanSchool of Health and Related Research (ScHARR), University of Sheffield, Regent Court, 30 Regent Street, Sheffield, S1 4DA, UK.ORCID 0000-0002-1025-312X

Funding

Department of Health DRF-2016-09-119Medical Research Council G0902159
6 · The paper itself

Abstract

BACKGROUND AND

objectiveThe extrapolation of estimated hazard functions can be an important part of cost-effectiveness analyses. Given limited follow-up time in the sample data, it may be expected that the uncertainty in estimates of hazards increases the further into the future they are extrapolated. The objective of this study was to illustrate how the choice of parametric survival model impacts on estimates of uncertainty about extrapolated hazard functions and lifetime mean survival.

methodsWe examined seven commonly used parametric survival models and described analytical expressions and approximation methods (delta and multivariate normal) for estimating uncertainty. We illustrate the multivariate normal method using case studies based on four representative hypothetical datasets reflecting hazard functions commonly encountered in clinical practice (constant, increasing, decreasing, or unimodal), along with a hypothetical cost-effectiveness analysis.

resultsDepending on the survival model chosen, the uncertainty in extrapolated hazard functions could be constant, increasing or decreasing over time for the case studies. Estimates of uncertainty in mean survival showed a large variation (up to sevenfold) for each case study. The magnitude of uncertainty in estimates of cost effectiveness, as measured using the incremental cost per quality-adjusted life-year gained, varied threefold across plausible models. Differences in estimates of uncertainty were observed even when models provided near-identical point estimates.

conclusionsSurvival model choice can have a significant impact on estimates of uncertainty of extrapolated hazard functions, mean survival and cost effectiveness, even when point estimates were similar. We provide good practice recommendations for analysts and decision makers, emphasizing the importance of considering the plausibility of estimates of uncertainty in the extrapolated period as a complementary part of the model selection process.

Indexed as

Clinical Trials as TopicCost-Benefit AnalysisModels, EconomicProportional Hazards ModelsHumansLikelihood FunctionsMonte Carlo MethodSurvival AnalysisUncertainty

Identifiers

PMID31761997
PMCPMC6976548

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LicenceCC BY
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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.