Cited by 600Statistical Methods in EpidemiologyMeta-analysis and systematic reviewsMethodologies in Health Research and Practice

The DOI matches its Crossref record; 83 of the 88 references it cites were found in a registry or an index; 2 of the 74 sources its claims cite were checked for support.

Reporting and Methods in Clinical Prediction Research: A Systematic Review

Open access

Abstract

BACKGROUND: We investigated the reporting and methods of prediction studies, focusing on aims, designs, participant selection, outcomes, predictors, statistical power, statistical methods, and predictive performance measures. METHODS AND FINDINGS: We used a full hand search to identify all prediction studies published in 2008 in six high impact general medical journals. We developed a comprehensive item list to systematically score conduct and reporting of the studies, based on recent recommendations for prediction research. Two reviewers independently scored the studies. We retrieved 71 papers for full text review: 51 were predictor finding studies, 14 were prediction model development studies, three addressed an external validation of a previously developed model, and three reported on a model's impact on participant outcome. Study design was unclear in 15% of studies, and a prospective cohort was used in most studies (60%). Descriptions of the participants and definitions of predictor and outcome were generally good. Despite many recommendations against doing so, continuous predictors were often dichotomized (32% of studies). The number of events per predictor as a measure of statistical power could not be determined in 67% of the studies; of the remainder, 53% had fewer than the commonly recommended value of ten events per predictor. Methods for a priori selection of candidate predictors were described in most studies (68%). A substantial number of studies relied on a p-value cut-off of p<0.05 to select predictors in the multivariable analyses (29%). Predictive model performance measures, i.e., calibration and discrimination, were reported in 12% and 27% of studies, respectively. CONCLUSIONS: The majority of prediction studies in high impact journals do not follow current methodological recommendations, limiting their reliability and applicability.

Overview

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TL;DR

This systematic review of 71 multivariable clinical prediction studies published in six high-impact general medical journals in 2008 found that most were predictor-finding studies and that key methodological and reporting recommendations were frequently not followed. Reporting was generally adequate for aims, participant selection, and predictor/outcome definitions, but poor for blinding, continuous predictors, power, missing data, multivariable result presentation, and model performance/validation.

Key findings

  • Of 1,204 hand-searched articles, 71 met inclusion criteria: 51 were predictor-finding studies, 14 were model development studies (11 without and 3 with external validation), 3 were external validation studies, and 3 were impact studies; these 71 papers studied 135 models or sets of predictors.
  • Statistical power was often inadequately reported: based on predictors in the final model, >10 events per variable occurred in 84 of 124 models, but candidate predictors were not described in the vast majority of studies; among studies clearly describing candidate predictors, effective sample size was <10 events per variable in 50% (n = 21) of presented models.
  • Missing data occurrence or handling was not described or unclear in 38% of all studies; complete case analysis was used in 65% (45/71), while multiple imputation was used in 7% (5/71) per Table 7 (the text states 8% of all studies).
  • Model performance and validation were limited: C-statistic/AUC-ROC was reported in 12% of predictor-finding and 80% of development/external-validation studies combined; calibration plots in 6% (4/66) and Hosmer-Lemeshow statistic in 9% (6/66); internal validation was performed in 33% (5/14) of model development studies, and external validation in only four studies.

Methods

  • Systematic review by hand search of the six highest-impact general medicine journals for 2008: *The New England Journal of Medicine*, *The Lancet*, *JAMA*, *Annals of Internal Medicine*, *BMJ*, and *PLoS Medicine*; original research with an abstract was eligible, and two reviewers independently assessed eligibility and extracted data, with median inter-reviewer agreement of 92% (IQR 75%–100%).
  • Included multivariable diagnostic and prognostic prediction studies predicting an outcome by two or more independent variables; excluded single predictor/test/marker studies, causality-only studies, and studies not contributing to patient care. An item list was developed from existing reporting statements/checklists (CONSORT, REMARK, STARD, STROBE, QUADAS) and prediction methodology recommendations.
  • Studies were classified into five types: predictor finding, model development without external validation, model development with external validation, external validation without or with model updating, and model impact; results were grouped by study type, medical specialty, and whether prediction was a primary or secondary aim.

Limitations

  • Stated: the review examined six high-impact journals, likely representing higher-quality studies; reporting may have improved since 2008, though the authors considered this unlikely because no major reporting guidelines for this research type had been produced recently; the work assessed reporting and statistical methods, not necessarily the appropriateness of design and conduct; conduct may be better than reported because journals limit paper length; poor reporting prevents readers from gauging relevance and reliability.
  • Stated: statistical power or effective sample size could not be assessed for many studies because of inadequate reporting of the number of candidate predictors; selective reporting of outcomes and predictors could not generally be assessed because study registration is not mandated for prediction research.
  • Evident from the text: the review is restricted to 2008 publications in six general medical journals, so findings may not generalize to other years, journals, or specialty journals; classification relied on full-text interpretation because authors generally did not explicitly state their aim, especially to distinguish predictor-finding from model-development studies.

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Integrity signals

Registry

Crossref · 10.1371/journal.pmed.1001221

Cites

83 of 83 references verified0 citation errors · 0 not checkable

Statistics

Nothing to recompute.

Figures

Figure 1 Flowchart of included studies. aThe hand search included only studies with an abstract, published in 2008 in The New England Journal of Medicine, The Lancet, JAMA: the Journal of the American Medical Association, Annals of Internal Medicine, BMJ, and PLoS Medicine. The following publication types were excluded beforehand: editorials, bibliographies, biographies, comments, dictionaries, directories, festschrifts, interviews, letters, news, and periodical indexes. bStudies, generally conducted in a yet healthy population, aimed at quantifying a causal relationship between a particular determinant or risk factor and an outcome, adjusting for other risk factors (i.e., confounders). cFor example, see [72].
Figure 1 Flowchart of included studies. aThe hand search included only studies with an abstract, published in 2008 in The New England Journal of Medicine, The Lancet, JAMA: the Journal of the American Medical Association, Annals of Internal Medicine, BMJ, and PLoS Medicine. The following publication types were excluded beforehand: editorials, bibliographies, biographies, comments, dictionaries, directories, festschrifts, interviews, letters, news, and periodical indexes. bStudies, generally conducted in a yet healthy population, aimed at quantifying a causal relationship between a particular determinant or risk factor and an outcome, adjusting for other risk factors (i.e., confounders). cFor example, see [72].CC BY · PubMed Central

Verification

Claims the paper makes

71 Own result · 26 Not checked yet · 12 No basis given · 5 Own interpretation · 4 Own method · 1 Partly supported · accuracy not yet measured

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  • It may state two separate claims as one. 37
  • Probably not a substantial claim. 13
47 of 47
  • The paper relies on this without support.

    Study registration is not mandated for prediction research.

    “study registration is not mandated for prediction research”
  • The authors state this as an assumption.

    The reason for missing data is not entirely due to non-observed information (data are not missing not at random) when multiple imputation is used.

    “on the assumption that the reason for the missing data is not entirely due to non-observed information (i.e., data are not “missing not at random”).”
  • The paper relies on this without support.

    The 71 prediction studies published in six high-impact general medical journals in 2008 are representative of current clinical prediction research.

    “We assessed the reporting and methods in all clinical prediction studies published in six high-impact general medical journals in 2008.”
    “We have described the state of current prediction research, and highlighted aspects that clearly need improvement.”
  • The paper relies on this without support.

    The statistical methods reported in the published prediction studies reflect the methods actually used in those studies.

    “We note that our work assessed researchers' reporting and statistical methods in modelling, and not necessarily the appropriateness of the design and conduct of the studies.”
4 of 4

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Citations

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Metadata

Authors
Bouwmeester, W., Zuithoff, N. P. A., Mallett, S., Geerlings, M. I., Vergouwe, Y., Steyerberg, E. W., Altman, D. G., & Moons, K. G. M.
Affiliations
  • University Medical Center Utrecht (NL)
  • University of Oxford (GB)
  • Erasmus MC (NL)
Corresponding author
Moons, Karel G. M.
Venue
PLoS Medicine
Published
2012-05-22
Volume
9
Issue
5
Pages
e1001221
ISSN
  • 1549-1277 (ISSN-L)
  • 1549-1676
OpenAlex
W1998392635
Cited by
600
References
88
Citations by year
2026: 24 · 2025: 39 · 2024: 36 · 2023: 47 · 2022: 65 · 2021: 50 · 2020: 65 · 2019: 36 · 2018: 40 · 2017: 43 · 2016: 40 · 2015: 46 · 2014: 38 · 2013: 24 · 2012: 7
FWCI
5.61
Type
journal article
Language
en
Open access
Locations
  • PLoS Medicine (publishedVersion, cc-by)
  • Europe PMC (PubMed Central) (publishedVersion, cc-by)
  • PubMed Central (publishedVersion, cc-by)
  • PubMed (publishedVersion)
  • EUR Research Repository (Erasmus University Rotterdam) (publishedVersion, other-oa)
  • EUR Research Repository (Erasmus University Rotterdam) (submittedVersion, other-oa)
  • RePub (Erasmus University Rotterdam) (submittedVersion, other-oa)
  • RePEc: Research Papers in Economics (submittedVersion)
  • DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
  • Figshare (submittedVersion, cc-by)
  • University of Birmingham Research Portal (University of Birmingham) (submittedVersion, other-oa)
Topics
  • Physical Sciences › Mathematics › Statistics and Probability › Statistical Methods in Epidemiology
  • Social Sciences › Decision Sciences › Statistics, Probability and Uncertainty › Meta-analysis and systematic reviews
  • Health Sciences › Nursing › Research and Theory › Methodologies in Health Research and Practice
Keywords
prediction model development, reporting quality, methodological quality, predictor selection, calibration, discrimination (statistics), external validation, prospective cohort, general medical journals
MeSH terms
Forecasting, Humans, Periodicals as Topic, Prospective Studies, Research Design, Biomedical Research, Research Report

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