Reporting and Methods in Clinical Prediction Research: A Systematic Review
Open accessAbstract
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.
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Written from the paper’s full text.
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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Claims the paper makes
0 of 74 cited sources checked · 0 supported · 0 flagged
- Findingown data
Study recruitment dates were reported in 88% of the studies.
“Study recruitment dates were reported in 88% of the studies.”
Quote in the paperNot a substantial claim - Findingreasoning
11/68 studies had all cause mortality as the outcome, where such bias would not be a factor.
“11/68 studies had all cause mortality as the outcome, where such bias would not be a factor.”
Quote in the paperNot a substantial claim - Findingcitation
Effective sample size was sufficient in the majority of the external validation studies (9/13 models).
“Given this, effective sample size was sufficient in the majority of the external validation studies (9/13 models).”
Quote in the paperNot a substantial claim - Findingown data
Outcomes were well defined in 62/68 studies.
“Outcomes were well defined in 62/68 studies (Table 3).”
Quote in the paperNot a substantial claim - Findingown data
A number of studies predicted a combined endpoint (14/71).
“A number of studies predicted a combined endpoint (14/71).”
Quote in the paperNot a substantial claim
- Findingown data
In the studies that clearly described the number of candidate predictors, the effective sample size was <10 events per variable in 50% (n = 21) of the presented models.
“In the studies that clearly described the number of candidate predictors, the effective sample size was <10 events per variable in 50% (n = 21) of the presented models.”
Quote in the paperSubstantial claim - Findingreasoning
Prediction of more than one outcome was very common in predictor finding studies.
“Prediction of more than one outcome was very common in predictor finding studies, apparently because of their exploratory aim (Table S1).”
Quote in the paperNot a substantial claim - Findingown data
There were few multivariable diagnostic studies (n = 5).
“There were few multivariable diagnostic studies (n = 5).”
Quote in the paperNot a substantial claim - Findingown data
Description of the cohort characteristics was clear in 68/69 of the relevant prediction studies.
“Description of the cohort characteristics was clear in 68/69 of the relevant prediction studies (not applicable for two non-nested case-control studies).”
Quote in the paperNot a substantial claim - Findingown data
Most multivariable prediction studies were published in the field of cardiovascular diseases (n = 24).
“Most multivariable prediction studies were published in the field of cardiovascular diseases (n = 24) (Table 1).”
Quote in the paperNot a substantial claim - Findingown data
The aim was usually to identify independently contributing predictors of an outcome (n = 51/71).
“The aim was usually to identify independently contributing predictors of an outcome (n = 51/71).”
Quote in the paperNot a substantial claim - Findingown data
Based on the number of predictors eventually included in the final model, statistical power was >10 events per variable in 84 of the 124 models fitted in predictor finding and model development studies.
“Based on the former, as expected, the statistical power was indeed >10 events per variable in 84 (11+60+1+13) of the 124 (96+28) models fitted in these studies.”
Quote in the paperSubstantial claim - Findingown data
Description of the candidate predictor variables was in general clear (59/68).
“Description of the candidate predictor variables was in general clear (59/68) (Table 4).”
Quote in the paperNot a substantial claim - Findingown data
Dichotomization of continuous predictors is still common practice (21/64).
“Dichotomization of continuous predictors is still common practice (21/64), despite being discouraged for decades [3],[50].”
Quote in the paperNot a substantial claim - Findingown data
Prospective cohort data, either observational or randomized trial data, were most frequently used (n = 44).
“Prospective cohort data, either observational or randomized trial data, were most frequently used (n = 44).”
Quote in the paperNot a substantial claim - Findingown data
In the 71 publications, 135 models or sets of predictors were studied.
“In the 71 publications, 135 models or sets of predictors were studied.”
Quote in the paperNot a substantial claim - Findingown data
In some studies, continuous, categorical, and time-to-event data were analyzed as binary outcomes.
“in some studies, continuous, categorical, and time-to-event data were analyzed as binary outcomes”
Quote in the paperSubstantial claim - Findingown data
Most studied outcomes were binary or time-to-event outcomes.
“Most studied outcomes were binary or time-to-event outcomes.”
Quote in the paperNot a substantial claim - Findingown data
Case-control designs were used only by predictor finding studies.
“We found that case-control designs were indeed used only by predictor finding studies (Table 2).”
Quote in the paperNot a substantial claim - Findingown data
Across all 71 included prediction studies, only 12 gave an explicit sample size calculation.
“Across all 71 included prediction studies, only 12 gave an explicit sample size calculation.”
Quote in the paperSubstantial claim - Findingnone given
It was difficult to distinguish, from study abstracts alone, between descriptive predictor finding studies and articles studying causality of a factor.
“It was difficult to distinguish, from study abstracts alone, between descriptive predictor finding studies and articles studying causality of a factor.”
Quote in the paperSubstantial claim - Findingown data
In 51 of the 68 studies, predictor measurement was blinded for the outcome.
“In 51 of the 68 studies, predictor measurement was blinded for the outcome: in 44, simply because of the prospective design; in seven non-prospective studies, predictor measurement was explicitly blinded for the outcome.”
Quote in the paperNot a substantial claim - Findingown data
Only 12 studies reported that they blinded the outcome measurement for predictor values.
“However, only 12 studies reported that they blinded the outcome measurement for predictor values.”
Quote in the paperNot a substantial claim - Findingown data
Participant recruitment was in general well described.
“Participant recruitment was in general well described.”
Quote in the paperNot a substantial claim - Findingown data
The majority of prediction studies published in 2008 in six high-impact general medical journals do not follow current methodological recommendations.
“Walter Bouwmeester and colleagues investigated the reporting and methods of prediction studies in 2008, in six high-impact general medical journals, and found that the majority of prediction studies do not follow current methodological recommendations.”
Quote in the paperSubstantial claim - Findingown data
Length of follow-up was not reported in nine studies.
“Length of follow-up was not reported in nine studies, leaving readers unable to know the time period for the predicted risks of the models.”
Quote in the paperNot a substantial claim - Findingown data
There was insufficient reporting of the number of candidate predictors in the vast majority of these studies, such that a proper estimation of the statistical power could not be made.
“However, there was insufficient reporting of the number of candidate predictors in the vast majority of these studies, such that a proper estimation of the statistical power could not be made.”
Quote in the paperSubstantial claim - Findingown data
Clinical setting was not reported in 4% of the studies.
“Clinical setting was not reported in 4% of the studies.”
Quote in the paperNot a substantial claim - Findingown data
Inclusion criteria were reported in 64/71 studies.
“Inclusion criteria were reported in 64/71 studies.”
Quote in the paperNot a substantial claim - Findingown data
The majority of studies involved participants from a hospital setting (38%) or the general (healthy) population (27%).
“The majority of studies involved participants from a hospital setting (38%) or the general (healthy) population (27%).”
Quote in the paperNot a substantial claim - Findingown data
The review found a similar number of model development studies (n = 14) and studies that aimed to evaluate an existing model (n = 6+3).
“However, we found a similar number of model development studies (n = 14), and studies that aimed to evaluate an existing model (n = 6+3).”
Quote in the paperNot a substantial claim - Findingown data
R2 and Brier score were reported in very few studies.
“R 2 and Brier score were reported in very few studies.”
Quote in the paperNot a substantial claim - Findingown data
The review could not assess statistical power or effective sample size for many studies because of inadequate reporting of the number of candidate predictors.
“Further, we could not assess statistical power or effective sample size for many studies because of inadequate reporting of the number of candidate predictors.”
Quote in the paperSubstantial claim - Findingown data
Calibration was reported in only a few studies.
“Calibration was reported in only a few studies (Table 9).”
Quote in the paperSubstantial claim - Findingown data
Occurrence and handling of missing values was not described or was unclear in 38% of all studies.
“Occurrence and handling of missing values was not described or was unclear in 38% of all studies (Table 7).”
Quote in the paperSubstantial claim - Findingown data
If missing values were reported, they were mostly reported by missing values per predictor (58%).
“If reported, it was mostly reported by “missing values per predictor” (58%).”
Quote in the paperNot a substantial claim - Findingown data
If calibration was done, the Hosmer-Lemeshow statistic was the most often reported calibration measure.
“If done, the Hosmer-Lemeshow statistic was the most often reported calibration measure.”
Quote in the paperNot a substantial claim - Findingown data
Other criteria, such as Akaike's Information Criterion or R 2, were used much less frequently.
“Other criteria, such as Akaike's Information Criterion or R 2, were used much less frequently.”
Quote in the paperNot a substantial claim - Findingown data
The method of selection of predictors within multivariable models was not described in 19% of the studies.
“The method of selection of predictors within multivariable models was not described in 19% of the studies.”
Quote in the paperNot a substantial claim - Findingown data
Few studies reported adjusted (20%) or unadjusted (18%) results of the full model with all candidate predictors considered.
“Few studies reported adjusted (20%) or unadjusted (18%) results of the full model with all candidate predictors considered (Table 8).”
Quote in the paperSubstantial claim - Findingown data
The missing indicator method was used in 13% of the studies investigated.
“it was still used in 13% of the studies investigated here.”
Quote in the paperNot a substantial claim - Findingcitation
Various basic items are well described and reported in prediction studies, for example aim, participant selection, inclusion and exclusion criteria, and design.
“We do acknowledge that various basic items are well described and reported in prediction studies, for example aim, participant selection, inclusion and exclusion criteria, and design.”
Quote in the paperNot a substantial claim - Findingown data
Loss to follow-up was reported in 46% of the studies where this was applicable.
“Loss to follow-up was reported in 46% of the studies where this was applicable.”
Quote in the paperNot a substantial claim - Findingreasoning
Flow diagrams were hardly ever presented in prediction studies.
“In contrast to randomized therapeutic trials, flow diagrams were hardly ever presented in prediction studies”
Quote in the paperSubstantial claim - Findingnone given
Most studies correctly reported the adjusted predictor effects derived from the final multivariable analyses.
“Most studies correctly reported the (adjusted) predictor effects derived from the final multivariable analyses.”
Quote in the paperSubstantial claim - Findingnone given
Univariable pre-selection and predictor selection in multivariable analyses based solely on p-values (with a <0.05 cut-off) was often used in predictor finding and model development studies.
“Univariable pre-selection and predictor selection in multivariable analyses based solely on p-values (with a <0.05 cut-off) was often used in predictor finding and model development studies.”
Quote in the paperSubstantial claim- Rests on an assumption: The statistical methods reported in the published prediction studies reflect the methods actually used in those studies.
- Findingown data
The majority of model development studies reported predictive performance in the development data only.
“The majority of model development studies reported predictive performance in the development data only.”
Quote in the paperSubstantial claim - Findingreasoning
Because reasons for missing values were insufficiently described in most studies that applied complete case analysis, it was impossible to judge whether imputation methods would have been appropriate.
“As the reasons for missing values were insufficiently described in most studies that applied a complete case analysis, it was impossible to judge whether imputation methods would indeed have been appropriate.”
Quote in the paperSubstantial claim - Findingown data
Several studies did not rely exclusively on p-value-based methods for predictor selection but also included established predictors in their model regardless of statistical significance in their dataset.
“Several studies, however, did not rely exclusively on these methods for predictor selection, but rather also, as is recommended, included established predictors in their model regardless of statistical significance in their dataset.”
Quote in the paperNot a substantial claim - Findingnone given
The exact number of events per candidate predictor could almost never be assessed.
“As the exact number of events per candidate predictor could almost never be assessed”
Quote in the paperSubstantial claim - Findingown data
The review observed much variation in the reporting of predictive performance measures.
“We observed much variation in the reporting of predictive performance measures.”
Quote in the paperSubstantial claim - Findingown data
The methods used for further predictor selection during the statistical analyses were poorly reported.
“However, the methods used for further predictor selection during the statistical analyses were poorly reported.”
Quote in the paperSubstantial claim- Rests on an assumption: The statistical methods reported in the published prediction studies reflect the methods actually used in those studies.
- Findingown data
Only a few studies also reported results of the univariable analyses.
“Only a few studies also reported results of the univariable analyses, which is often a useful comparator.”
Quote in the paperNot a substantial claim - Findingcitation
Good reporting of how candidate predictors were pre-selected in this review compares favourably with other reviews.
“Good reporting of how candidate predictors were pre-selected in our review compares favourably with other reviews [15],[20],[21],[29],[33],[35],[36],[48].”
Quote in the paperNot a substantial claim0 of 3 sources checked- cites [15] A Laupacis, 1997, Clinical prediction rules. a review and suggested modifications of methodological standards., JAMA source
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- cites [20] S Mallett, 2010, Reporting performance of prognostic models in cancer: a review., BMC Med source
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- cites [21] S Mallett, 2010, Reporting methods in studies developing prognostic models in cancer: a review., BMC Med source
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- Findingreasoning
It was not possible to determine whether reported results were subject to overfitting or optimistic predictive performances.
“it was not possible to determine whether reported results were indeed subject to overfitting or optimistic predictive performances.”
Quote in the paperSubstantial claim - Premisecitation
The use of a combined endpoint will give problems if the predictor effect is in opposite directions for different outcomes included in the composite endpoint.
“The use of a combined endpoint will give problems if the predictor effect is in opposite directions for different outcomes included in the composite endpoint [52],[53].”
Quote in the paperNot a substantial claim0 of 2 sources checked - Premisecitation
Knowledge of the predictors might influence outcome assessment, resulting in a biased estimation of the predictor effects for the outcome.
“Knowledge of the predictors might influence outcome assessment, resulting in a biased estimation of the predictor effects for the outcome [6],[17],[39].”
Quote in the paperSubstantial claim0 of 3 sources checked- cites [6] KG Moons, 2009, Prognosis and prognostic research: what, why, and how?, BMJ source
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- cites [17] JG Lijmer, 1999, Empirical evidence of design-related bias in studies of diagnostic tests., JAMA source
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- cites [39] PM Bossuyt, 2003, Towards complete and accurate reporting of studies of diagnostic accuracy: The STARD Initiative., Ann Intern Med source
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- Premisecitation
Use of poor methods may lead to biased results.
“Use of poor methods may lead to biased results [2],[7],[10],[12],[13],[15],[17]–[32].”
Quote in the paperSubstantial claim0 of 3 sources checked- cites [2] DG Altman, 2007, Prognostic models: a methodological framework and review of models for breast cancer.
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- cites [7] P Royston, 2009, Prognosis and prognostic research: developing a prognostic model., BMJ source
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- cites [10] EW Steyerberg, 2009, Clinical prediction models: a practical approach to development, validation, and updating source
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- Premisecitation
Selective reporting of outcomes (and predictors) is often a risk.
“However, selective reporting of outcomes (and predictors) is often a risk [51].”
Quote in the paperSubstantial claim0 of 1 sources checked- cites [51] JJ Kirkham, 2010, The impact of outcome reporting bias in randomised controlled trials on a cohort of systematic reviews., BMJ source
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- Premisecitation
Quantifying the impact of a prediction model on participant outcome requires a comparative study design.
“Quantifying the impact of a prediction model on participant outcome requires a comparative study design [9],[48].”
Quote in the paperNot a substantial claim0 of 2 sources checked- cites [9] KG Moons, 2009, Prognosis and prognostic research: application and impact of prognostic models in clinical practice., BMJ source
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- cites [48] BM Reilly, 2006, Translating clinical research into clinical practice: impact of using prediction rules to make decisions., Ann Intern Med source
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- Premisecitation
For time-to-event outcomes, the effective sample size is also highly related to the number of participants who experience the event.
“For time-to-event outcomes, the effective sample size is also highly related to the number of participants who experience the event [12].”
Quote in the paperNot a substantial claim0 of 1 sources checked- cites [12] FE Harrell, 2001, Regression modeling strategies with applications to linear models, logistic regression and survival analysis
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- Premisecitation
The use of multivariable selection methods can also lead to overfitting and unstable models, especially when there are relatively few outcome events and many predictors analysed.
“However, the use of multivariable selection methods can also lead to overfitting and unstable models, especially when there are relatively few outcome events and many predictors analysed [10],[12],[13].”
Quote in the paperSubstantial claim0 of 3 sources checked- cites [10] EW Steyerberg, 2009, Clinical prediction models: a practical approach to development, validation, and updating source
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- cites [12] FE Harrell, 2001, Regression modeling strategies with applications to linear models, logistic regression and survival analysis
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- cites [13] FE Harrell Jr, 1996, Multivariable prognostic models: issues in developing models, evaluating assumptions and adequacy, and measuring and reducing errors., Stat Med source
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- Interpretationreasoning
It is generally impossible to assess whether some outcomes were analysed but not reported in prediction research because study registration is not mandated.
“Unfortunately, study registration is not mandated for prediction research, so it is generally impossible to assess whether some outcomes were analysed but not reported.”
Quote in the paperSubstantial claim- Rests on an assumption: Study registration is not mandated for prediction research.
- Premisecitation
Most guidelines advise investigators to report both unadjusted and adjusted results.
“Most guidelines, such as the STROBE guidelines for the reporting of observational studies or the REMARK guidelines for tumour marker prognostic studies, specifically advise investigators to report both unadjusted results (i.e., from univariable analysis, yielding the association of each candidate predictor with the outcome) and adjusted results (i.e., from a multivariable analysis) [4],[38],[63].”
Quote in the paperSubstantial claim0 of 3 sources checked- cites [4] LM McShane, 2005, Reporting recommendations for tumor marker prognostic studies (REMARK)., J Natl Cancer Inst source
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- cites [38] E Von Elm, 2007, The Strengthening the Reporting of Observational Studies in Epidemiology (STROBE) statement: guidelines for reporting observational studies., Lancet source
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- cites [63] JP Vandenbroucke, 2007, Strengthening the Reporting of Observational Studies in Epidemiology (STROBE): explanation and elaboration., PLoS Med source
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- Premisecitation
Missing values in clinical study data rarely occur completely at random.
“Missing values in clinical study data rarely occur completely at random.”
Quote in the paperSubstantial claim0 of 7 sources checked- cites [10] EW Steyerberg, 2009, Clinical prediction models: a practical approach to development, validation, and updating source
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- cites [12] FE Harrell, 2001, Regression modeling strategies with applications to linear models, logistic regression and survival analysis
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- cites [13] FE Harrell Jr, 1996, Multivariable prognostic models: issues in developing models, evaluating assumptions and adequacy, and measuring and reducing errors., Stat Med source
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- cites [23] AR Donders, 2006, Review: a gentle introduction to imputation of missing values., J Clin Epidemiol source
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- cites [24] S Greenland, 1995, A critical look at methods for handling missing covariates in epidemiologic regression analyses., Am J Epidemiol source
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- cites [57] A Burton, 2004, Missing covariate data within cancer prognostic studies: a review of current reporting and proposed guidelines., Br J Cancer source
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- cites [58] MH Gorelick, 2006, Bias arising from missing data in predictive models., J Clin Epidemiol source
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- Premisecitation
Commonly missing values are related to observed participant or disease characteristics.
“Commonly missing values are related to observed participant or disease characteristics.”
Quote in the paperSubstantial claim0 of 7 sources checked- cites [10] EW Steyerberg, 2009, Clinical prediction models: a practical approach to development, validation, and updating source
Not checked in the free run.
- cites [12] FE Harrell, 2001, Regression modeling strategies with applications to linear models, logistic regression and survival analysis
Not checked in the free run.
- cites [13] FE Harrell Jr, 1996, Multivariable prognostic models: issues in developing models, evaluating assumptions and adequacy, and measuring and reducing errors., Stat Med source
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- cites [23] AR Donders, 2006, Review: a gentle introduction to imputation of missing values., J Clin Epidemiol source
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- cites [24] S Greenland, 1995, A critical look at methods for handling missing covariates in epidemiologic regression analyses., Am J Epidemiol source
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- cites [57] A Burton, 2004, Missing covariate data within cancer prognostic studies: a review of current reporting and proposed guidelines., Br J Cancer source
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- cites [58] MH Gorelick, 2006, Bias arising from missing data in predictive models., J Clin Epidemiol source
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- Interpretationreasoning
It is likely that studies that did not or unclearly reported the method of handling missing values applied a complete case analysis as well.
“It is likely that the studies that did not or unclearly reported the method of handling missing values applied a complete case analysis as well.”
Quote in the paperNot a substantial claim - Premisecitation
The missing indicator method is known to lead to biased results in almost all observational studies.
“Even though this method is known to lead to biased results in almost all observational studies [13],[23],[60]–[62]”
Quote in the paperNot a substantial claim0 of 3 sources checked- cites [13] FE Harrell Jr, 1996, Multivariable prognostic models: issues in developing models, evaluating assumptions and adequacy, and measuring and reducing errors., Stat Med source
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- cites [23] AR Donders, 2006, Review: a gentle introduction to imputation of missing values., J Clin Epidemiol source
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- cites [60]–[62]
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- Premisecitation
Overfitting is even more likely when the number of candidate predictors is large relative to the effective sample size.
“Overfitting is even more likely when the number of candidate predictors is large relative to the effective sample size [10],[12],[70].”
Quote in the paperNot a substantial claim0 of 3 sources checked- cites [10] EW Steyerberg, 2009, Clinical prediction models: a practical approach to development, validation, and updating source
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- cites [12] FE Harrell, 2001, Regression modeling strategies with applications to linear models, logistic regression and survival analysis
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- cites [70] JC Van Houwelingen, 1990, Predictive value of statistical models., Stat Med source
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- Premisecitation
The extent of optimism in apparent model performance may be estimated with internal validation techniques.
“The extent of this optimism may be estimated with so-called internal validation techniques [10],[12],[43],[70]”
Quote in the paperSubstantial claim0 of 3 sources checked- cites [10] EW Steyerberg, 2009, Clinical prediction models: a practical approach to development, validation, and updating source
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- cites [12] FE Harrell, 2001, Regression modeling strategies with applications to linear models, logistic regression and survival analysis
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- cites [43] EW Steyerberg, 2001, Internal validation of predictive models: efficiency of some procedures for logistic regression analysis., J Clin Epidemiol source
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- Interpretationreasoning
Predictor selection based solely on p-values may be problematic with low numbers of events and many candidate predictors.
“This approach may notably be problematic with low numbers of events and many candidate predictors.”
Quote in the paperSubstantial claim - Interpretationreasoning
The generalisability of the performance of the reported models, especially in studies where prediction was the primary aim, is difficult to evaluate.
“Accordingly, the generalisability of the performance of these reported models, especially in studies where prediction was the primary aim, is difficult to evaluate.”
Quote in the paperSubstantial claim
- May combine two claims. 37
- Classified as probably not a substantial claim. 13
May combine two claims.
Findingown dataA randomized trial was used by two of the three impact studies; the third used an observational before-after (prospective) cohort design.
“A randomized trial was used by two of the three impact studies; the third used an observational before-after (prospective) cohort design, comparing participant outcomes before and after the introduction of a prediction model.”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataOf the prediction modelling studies (n = 20), the vast majority were model development studies, without (n = 11) or with (n = 3) external validation.
“Of the prediction modelling studies (n = 20), the vast majority were model development studies, without (n = 11) or with (n = 3) external validation.”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataPredictor interaction (non-additivity) was tested in 25 of the 51 predictor finding studies and in 11 of the 14 model development studies (total n = 36).
“Predictor interaction (non-additivity) was tested in 25 of the 51 predictor finding studies and in 11 of the 14 model development studies (total n = 36).”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataPure external validation (n = 3) and impact studies (n = 3) were rare.
“Pure external validation (n = 3) and impact studies (n = 3) were rare.”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataWhether (all) consecutive participants were included or how many participants refused to participate was rarely reported and so could not be evaluated.
“Whether (all) consecutive participants were included or how many participants refused to participate was rarely reported and so could not be evaluated.”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataIn outcome reporting, differences between studies with prediction as a primary versus a secondary aim were expected but not observed.
“In outcome reporting, we expected differences between studies with prediction as a primary versus a secondary aim, but this was not observed.”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataHand searching identified 1,204 articles, of which 71 met the inclusion criteria.
“We identified 1,204 articles by hand searching, of which 71 met the inclusion criteria (Figure 1 and Text S2).”
Quote in the paperSubstantial claimProbably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.
Findingown dataAuthors in general did not explicitly state their aim, so full text interpretations were used to classify studies as predictor finding or model development studies.
“Authors in general did not explicitly state their aim, so we used full text interpretations to classify studies as predictor finding or model development studies.”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataDiscrimination was assessed with the C-statistic or area under the ROC curve in 12% of predictor finding studies and 80% of model development and external validation studies.
“Discrimination was assessed with the C-statistic or area under the receiver operating characteristic (ROC) curve in 12% of the predictor finding and, as expected, 80% of the model development and external validation studies.”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataThe majority of predictor finding studies (65%) and model development studies (79%) reported predictor coefficients or effect estimates of the final model after predictor selection.
“The majority, 65% of the predictor finding and 79% of the model development studies, reported the predictor coefficients or effect estimates of the model after predictor selection (the final model).”
Quote in the paperSubstantial claimMay combine two claims.
Findingown dataMultiple imputation was used in only 8% of all studies and was applied and reported in very few studies.
“By comparison, multiple imputation, the most rigorous strategy for dealing with missing values, was used in only 8% of all studies.”
- Also stated in: § Discussion · hedged
“Recommended methods, such as multiple imputation, were applied and reported in very few studies, although this may be due to the fact that consensus in recommending these methods was arrived at only recently.”
Quote in the paperSubstantial claim- Also stated in: § Discussion · hedged
May combine two claims.
Findingnone givenIn descriptions of participant selection, it often remained unclear whether participants were included in an unbiased way, notably with respect to refusals and whether all consecutive eligible participants were included.
“In descriptions of participant selection, it often remained unclear whether participants were included in an unbiased way, notably with respect to refusals and whether all consecutive eligible participants were included.”
Quote in the paperSubstantial claimMay combine two claims.
Findingown dataExternal validation was performed in only four studies, and only a very few model development studies reported an external validation of the model in the same paper.
“and external validation in only four studies.”
- Also stated in: § Discussion · plain
“Similarly, only a very few model development studies reported an external validation of the model in the same paper.”
Quote in the paperSubstantial claim- Also stated in: § Discussion · plain
May combine two claims.
Findingown dataExternal validation and model impact studies were rare (n = 6), and only a very small minority of papers involved external validations or impact assessments of a previously developed prediction model.
“External validation and model impact studies were rare (n = 6).”
- Also stated in: § Discussion · plain
“Only a very small minority of the papers involved the most useful approaches in predicting participant clinical outcomes, namely, external validations or impact assessments of a previously developed prediction model.”
Quote in the paperSubstantial claim- Also stated in: § Discussion · plain
May combine two claims.
Findingown dataStudy design, participant selection, definitions of outcomes and predictors, and predictor selection were generally well reported.
“Study design, participant selection, definitions of outcomes and predictors, and predictor selection were generally well reported.”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown data18% of all studies investigated the added value of specific predictors, and 42% included predictors regardless of statistical significance.
“18% of all studies investigated the added value of specific predictors, and 42% included predictors regardless of statistical significance.”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataComplete case analysis was performed in the vast majority of studies and was by far the most commonly used approach to handle missing values.
“Analysis of participants with complete data (i.e., complete case analysis) was performed in the vast majority of studies.”
- Also stated in: § Discussion · plain
“Complete case analysis was by far the most commonly used approach to handle missing values, despite many methodological recommendations to do otherwise [10],[12],[13],[23],[24],[57],[58].”
Quote in the paperSubstantial claim- Also stated in: § Discussion · plain
May combine two claims.
Findingown dataThe most commonly reported criterion for predictor selection in multivariable models was a p-value of <0.05, used in 18% of all studies (and in 26% of the studies that indeed applied predictor selection in multivariable analyses).
“The most commonly reported criterion for predictor selection in multivariable models was a p-value of <0.05, used in 18% of all studies (and in 26% of the studies that indeed applied predictor selection in multivariable analyses).”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataAmong the 71 prediction studies identified, the vast majority were predictor finding studies (n = 51), followed by model development studies (n = 14).
“Our investigation found that among the 71 prediction studies identified, the vast majority were predictor finding studies (n = 51), followed by model development studies (n = 14).”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataFourteen studies developed new prediction models, of which three included an external validation.
“We found that 14 studies developed new prediction models (of which three included an external validation).”
Quote in the paperSubstantial claimMay combine two claims.
Findingown dataThree studies externally validated models, and three investigated the impact of an existing prediction model.
“Three studies externally validated models, and three investigated the impact of an existing prediction model.”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataStudies reported using backward selection in 17% of the primary-aim and 18% of the secondary-aim studies, whereas forward selection was reported in 6% and 0%, respectively.
“Studies reported using backward selection in 17% of the primary-aim and 18% of the secondary-aim studies, whereas forward selection was reported in 6% and 0%, respectively.”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataIn studies with prediction as the primary aim, the majority (71%) selected their candidate predictors based on existing literature, whereas this was less often the case (29%) in studies with prediction as a secondary aim.
“In studies with prediction as the primary aim, the majority (71%) selected their candidate predictors based on existing literature, whereas this was less often the case (29%) in studies with prediction as a secondary aim.”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataInternal validation was performed in 33% (n = 5) of the 14 model development studies, and use of internal validation techniques was rare.
“Internal validation was performed in 33% (n = 5) of the 14 model development studies”
- Also stated in: § Discussion · plain
“but use of these techniques was rare.”
Quote in the paperSubstantial claim- Also stated in: § Discussion · plain
May combine two claims.
Findingown dataPre-selection of candidate predictors for inclusion in the multivariable analyses based on univariable predictor-outcome associations was used in 13% of the primary-aim and in 24% of the secondary-aim prediction studies.
“Pre-selection of candidate predictors for inclusion in the multivariable analyses based on univariable predictor–outcome associations was used in 13% of the primary-aim and in 24% of the secondary-aim prediction studies.”
Quote in the paperNot a substantial claimMay combine two claims.
Findingown dataSelection of candidate predictors was described for 36 (75%) of the studies where prediction was the primary aim and for eight (47%) of the studies where prediction was a secondary aim.
“We found that selection of candidate predictors was described for 36 (75%) of the studies where prediction was the primary aim and for eight (47%) of the studies where prediction was a secondary aim (Table 6).”
Quote in the paperSubstantial claimMay combine two claims.
Findingown dataSample size considerations, applied statistical methods, and procedures for dealing with missing data were poorly reported despite being highlighted in several reporting guidelines.
“However, sample size considerations, applied statistical methods, and procedures for dealing with missing data were poorly reported despite being highlighted in several reporting guidelines.”
Quote in the paperSubstantial claim- Rests on an assumption: The statistical methods reported in the published prediction studies reflect the methods actually used in those studies.
May combine two claims.
Findingnone givenMost studies reported the occurrence of missing data but did not report sufficient detail.
“Most studies reported the occurrence of missing data but did not report sufficient detail.”
Quote in the paperSubstantial claimMay combine two claims.
Findingown dataC-statistics or area under the ROC curves were the most frequently reported discrimination statistics, whereas measures of calibration and overall measures of fit were rarely reported.
“C-statistics or area under the ROC curves were the most frequently reported discrimination statistics, whereas measures of calibration (such as calibration slope) and overall measures of fit were rarely reported.”
Quote in the paperSubstantial claimMay combine two claims.
Findingown dataEncouraging findings included the frequent use of prospective studies, and adequate description of participant selection, predictor and outcome definitions, and the process for (pre)selection of candidate predictors.
“However, encouraging findings included the frequent use of prospective studies, and adequate description of participant selection, predictor and outcome definitions, and the process for (pre)selection of candidate predictors.”
Quote in the paperSubstantial claimProbably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.
Study design and conductown dataTwo authors independently extracted data, and in case of doubt items were discussed with a third and fourth reviewer.
“Two authors (W. B., N. P. A. Z.) independently extracted the data. In case of doubt, items were discussed with a third and fourth reviewer (K. G. M. M., Y. V.).”
Quote in the paperNot a substantial claimMay combine two claims.
PremisecitationSelection based on (significant) univariable predictor-outcome associations is not recommended, as this method increases the chance of biased results in terms of spurious predictors and overfitted and unstable models.
“However, many methodological reports have shown that selection based on (significant) univariable predictor–outcome associations is not recommended, as this method increases the chance of biased results in terms of spurious predictors and overfitted and unstable models [12],[13],[43],[56].”
Quote in the paperSubstantial claim0 of 3 sources checked- cites [12] FE Harrell, 2001, Regression modeling strategies with applications to linear models, logistic regression and survival analysis
Not checked in the free run.
- cites [13] FE Harrell Jr, 1996, Multivariable prognostic models: issues in developing models, evaluating assumptions and adequacy, and measuring and reducing errors., Stat Med source
Not checked in the free run.
- cites [43] EW Steyerberg, 2001, Internal validation of predictive models: efficiency of some procedures for logistic regression analysis., J Clin Epidemiol source
Not checked in the free run.
Classified as probably not a substantial claim.
PremisecitationTo externally validate a prediction model, a minimum effective sample size of 100 participants with and 100 without the event has been recommended.
“To externally validate a prediction model, a minimum effective sample size of 100 participants with and 100 without the event has been recommended [55].”
Quote in the paperSubstantial claim0 of 1 sources checked- cites [55] Y Vergouwe, 2005, Substantial effective sample sizes were required for external validation studies of predictive logistic regression models., J Clin Epidemiol source
Not checked in the free run.
Probably not a substantial claim: may not pass S3 (load-bearing).
PremisecitationA frequently mentioned rule of thumb is 10 events needed per candidate predictor.
“A frequently mentioned rule of thumb is “10 events needed per candidate predictor” [12],[25],[26],[30],[54].”
Quote in the paperNot a substantial claim0 of 3 sources checked- cites [12] FE Harrell, 2001, Regression modeling strategies with applications to linear models, logistic regression and survival analysis
Not checked in the free run.
- cites [25] P Peduzzi, 1996, A simulation study of the number of events per variable in logistic regression analysis., J Clin Epidemiol source
Not checked in the free run.
- cites [26] P Peduzzi, 1995, Importance of events per independent variable in proportional hazards regression analysis. II. Accuracy and precision of regression estimates., J Clin Epidemiol source
Not checked in the free run.
Classified as probably not a substantial claim.
PremisecitationAnalyzing continuous, categorical, and time-to-event data as binary outcomes is not recommended because less accurate predictions are likely to result.
“a practice that is not recommended because less accurate predictions are likely to result, as with dichotomizing predictor variables [50].”
Quote in the paperSubstantial claim0 of 1 sources checked- cites [50] P Royston, 2006, Dichotomizing continuous predictors in multiple regression: a bad idea., Stat Med source
Not checked in the free run.
May combine two claims.
PremisecitationAdequate reporting of predictor selection methods used is important, because the number of candidate predictors and how they were selected can influence the specific predictors included in the final multivariable model and affect interpretation of results.
“Adequate reporting of predictor selection methods used is important, because the number of candidate predictors and how they were selected at various stages of the study can both influence the specific predictors included in the final multivariable model, and thus affect the interpretation of the results 12,13,43,56.”
Quote in the paperSubstantial claim0 of 3 sources checked- cites [12] FE Harrell, 2001, Regression modeling strategies with applications to linear models, logistic regression and survival analysis
Not checked in the free run.
- cites [13] FE Harrell Jr, 1996, Multivariable prognostic models: issues in developing models, evaluating assumptions and adequacy, and measuring and reducing errors., Stat Med source
Not checked in the free run.
- cites [43] EW Steyerberg, 2001, Internal validation of predictive models: efficiency of some procedures for logistic regression analysis., J Clin Epidemiol source
Not checked in the free run.
Probably not a substantial claim: may not pass S3 (load-bearing).
Study design and conductown dataThe two reviewers agreed on a median of 92% (interquartile range 75%-100%) of the items extracted.
“The two reviewers agreed on a median of 92% (interquartile range 75%–100%) of the items extracted.”
Quote in the paperNot a substantial claimProbably not a substantial claim: may not pass S3 (load-bearing). May combine two claims.
Study design and conductown dataTwo authors independently assessed eligibility of studies, and a third independent reader was involved in case of doubt.
“The full text of all thus selected studies was obtained, and two authors (W. B. and N. P. A. Z.) independently assessed eligibility; in case of doubt a third independent reader was involved (K. G. M. M. or Y. V.).”
Quote in the paperNot a substantial claimProbably not a substantial claim: may not pass S3 (load-bearing).
Study design and conductnone givenInter-reviewer agreement on data extraction was assessed by calculating the percentage of overall agreement between the two reviewers.
“The inter-reviewer agreement on the data extraction was assessed by calculating the percentage of overall agreement between the two reviewers.”
Quote in the paperNot a substantial claimProbably not a substantial claim: may not pass S3 (load-bearing).
PremisereasoningMultiple imputation is often advocated to preserve power and obtain less biased results, assuming the reason for missing data is not entirely due to non-observed information (data are not missing not at random).
“Imputation, notably multiple imputation, of missing values is often advocated to preserve power and obtain less biased results, 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”).”
Quote in the paperNot a substantial claim- 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.
Probably not a substantial claim: may not pass S3 (load-bearing).
PremisecitationVarious reports have indicated that in prediction modelling research there is an unfortunate practice of developing new models instead of externally validating or updating existing models.
“Various reports have indicated that in prediction modelling research there is an unfortunate practice of developing new models instead of externally validating or updating existing models [8]–[10],[15],[29],[36],[48].”
Quote in the paperNot a substantial claim0 of 3 sources checked- cites [8]–[10]
Not checked in the free run.
- cites [15] A Laupacis, 1997, Clinical prediction rules. a review and suggested modifications of methodological standards., JAMA source
Not checked in the free run.
- cites [29] JH Wasson, 1985, Clinical prediction rules. Applications and methodological standards., N Engl J Med source
Not checked in the free run.
May combine two claims.
PremisecitationExclusion of participants with missing values often leads to loss of statistical power and biased results.
“Exclusion of participants with missing values will therefore not only lead to loss of statistical power, but often also to biased results 10,12,13,23,24,57,58.”
Quote in the paperSubstantial claim0 of 7 sources checked- cites [10] EW Steyerberg, 2009, Clinical prediction models: a practical approach to development, validation, and updating source
Not checked in the free run.
- cites [12] FE Harrell, 2001, Regression modeling strategies with applications to linear models, logistic regression and survival analysis
Not checked in the free run.
- cites [13] FE Harrell Jr, 1996, Multivariable prognostic models: issues in developing models, evaluating assumptions and adequacy, and measuring and reducing errors., Stat Med source
Not checked in the free run.
- cites [23] AR Donders, 2006, Review: a gentle introduction to imputation of missing values., J Clin Epidemiol source
Not checked in the free run.
- cites [24] S Greenland, 1995, A critical look at methods for handling missing covariates in epidemiologic regression analyses., Am J Epidemiol source
Not checked in the free run.
- cites [57] A Burton, 2004, Missing covariate data within cancer prognostic studies: a review of current reporting and proposed guidelines., Br J Cancer source
Not checked in the free run.
- cites [58] MH Gorelick, 2006, Bias arising from missing data in predictive models., J Clin Epidemiol source
Not checked in the free run.
Probably not a substantial claim: may not pass S3 (load-bearing).
PremisecitationThese basic items have been identified as important in several well-known guidelines for reporting of clinical research.
“These items have been identified as important in several well-known guidelines for reporting of clinical research [4],[38]–[41].”
Quote in the paperNot a substantial claim0 of 2 sources checked- cites [4] LM McShane, 2005, Reporting recommendations for tumor marker prognostic studies (REMARK)., J Natl Cancer Inst source
Not checked in the free run.
- cites [38]–[41]
Not checked in the free run.
Probably not a substantial claim: may not pass S3 (load-bearing).
PremisecitationPoor reporting of sample size rationale has also been observed by others.
“Poor reporting of sample size rationale has also been observed by others [3],[21],[68].”
Quote in the paperNot a substantial claim0 of 3 sources checked- cites [3] DG Altman, 1998, Methodological challenges in the evaluation of prognostic factors in breast cancer., Breast Cancer Res Treat source
Not checked in the free run.
- cites [21] S Mallett, 2010, Reporting methods in studies developing prognostic models in cancer: a review., BMC Med source
Not checked in the free run.
- cites [68] LM Bachmann, 2006, Sample sizes of studies on diagnostic accuracy: literature survey., BMJ source
Not checked in the free run.
May combine two claims.
Interpretationown dataThe review identified poor reporting and poor methods in many published prediction studies, which limits the reliability and applicability of the published findings.
“To conclude, we identified poor reporting and poor methods in many published prediction studies, which limits the reliability and applicability of the published findings.”
Quote in the paperSubstantial claim- Rests on an assumption: The 71 prediction studies published in six high-impact general medical journals in 2008 are representative of current clinical prediction research.
- Rests on an assumption: The statistical methods reported in the published prediction studies reflect the methods actually used in those studies.
May combine two claims.
PremisereasoningApparent model performance is generally too optimistic, as the model has been tailored to the dataset at hand.
“This apparent model performance, however, is generally too optimistic, as the model has been tailored to the dataset at hand.”
Quote in the paperSubstantial claim0 of 3 sources checked- cites [10] EW Steyerberg, 2009, Clinical prediction models: a practical approach to development, validation, and updating source
Not checked in the free run.
- cites [12] FE Harrell, 2001, Regression modeling strategies with applications to linear models, logistic regression and survival analysis
Not checked in the free run.
- cites [70] JC Van Houwelingen, 1990, Predictive value of statistical models., Stat Med source
Not checked in the free run.
Probably not a substantial claim: may not pass S3 (load-bearing).
PremisecitationThe REMARK guidelines for prognostic tumour marker studies recommend using a REMARK profile table instead of a flow diagram.
“The REMARK guidelines for prognostic tumour marker studies recommend using a REMARK profile table instead of a flow diagram [69].”
Quote in the paperNot a substantial claim0 of 1 sources checked- cites [69] S Mallett, 2010, Reporting of prognostic studies of tumour markers: a review of published articles in relation to REMARK guidelines., Br J Cancer source
Not checked in the free run.
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.”
Claims other papers make about this one
Each is checked against this paper’s own text: does it say what the citing paper says it does?
In 71 reports published in 2008 in 6 high-impact general medical journals, the level of reporting was overwhelmingly poor.
“Bouwmeester and colleagues [23] evaluated 71 reports, published in 2008 in 6 high-impact general medical journals, and likewise observed an overwhelmingly poor level of reporting.”
From Transparent reporting of a multivariable prediction model for individual prognosis or diagnosis (TRIPOD): the TRIPOD Statement (2015) · cites this paper as [23]
The quality of reporting in published articles describing the development or validation of multivariable prediction models in medicine is poor.
“Many reviews have showed that the quality of reporting in published articles describing the development or validation of multivariable prediction models in medicine is poor [23–27,29].”
From Transparent reporting of a multivariable prediction model for individual prognosis or diagnosis (TRIPOD): the TRIPOD Statement (2015) · cites this paper as [23]
Extraction of calibration measures is often hampered by poor assessment and reporting.
“Unfortunately, extraction of calibration measures is often hampered by poor assessment and reporting.11,16”
From A framework for meta-analysis of prediction model studies with binary and time-to-event outcomes (2019) · cites this paper as [16]
In meta-analysis without individual participant data, performance measures must be extracted from study publications, but they are often poorly reported.
“In a meta-analysis without access to individual participant data (IPD), we are reliant on extracting these performance measures from study publications. However, they are often poorly reported.”
From A framework for meta-analysis of prediction model studies with binary and time-to-event outcomes (2019) · cites this paper as [16]
Validation studies rarely present information on the calibration intercept and slope, or different studies report estimates for different risk strata or time horizons.
“For instance, validation studies rarely present information on the calibration intercept and slope, or different studies report estimates for different risk strata or time horizons.”
From A framework for meta-analysis of prediction model studies with binary and time-to-event outcomes (2019) · cites this paper as [16]
The quality of reporting of prediction model studies is generally poor.
“However, the overwhelming evidence shows that the quality of reporting of prediction model studies is poor.”
From Transparent Reporting of a Multivariable Prediction Model for Individual Prognosis or Diagnosis (TRIPOD): The TRIPOD Statement (2015) · cites this paper as [23]
Bouwmeester and colleagues evaluated 71 reports published in 2008 in 6 high-impact general medical journals and observed an overwhelmingly poor level of reporting.
“Bouwmeester and colleagues23 evaluated 71 reports, published in 2008 in 6 high-impact general medical journals, and likewise observed an overwhelmingly poor level of reporting.”
From Transparent Reporting of a Multivariable Prediction Model for Individual Prognosis or Diagnosis (TRIPOD): The TRIPOD Statement (2015) · cites this paper as [23]
The quality of reporting in published articles describing the development or validation of multivariable prediction models in medicine is poor.
“Many reviews have showed that the quality of reporting in published articles describing the development or validation of multivariable prediction models in medicine is poor.23–27,29”
From Transparent Reporting of a Multivariable Prediction Model for Individual Prognosis or Diagnosis (TRIPOD): The TRIPOD Statement (2015) · cites this paper as [23]
Self-check
Percentages, sums and reported differences re-computed from the paper’s own numbers.
Each contradiction between two passages listed here was confirmed by an independent check.
Predictor-combination total does not equal subgroup counts
MinorTable 6 lists eight primary-aim and one secondary-aim study combining similar predictors, but reports a total of seven rather than nine.
Reported 7 · recomputed 9
“| Similar predictors combinedd | 17 (8) | 6 (1) | 11 (7) |”
Missing-data reporting percentage does not match its count
MinorTable 7 reports 18 of 48 primary-aim studies as not reporting missing data clearly. That is 37.5%, not 35%.
Reported 35 · recomputed 37.5
“| Not reported or unclear | 35 (18) | 53 (9) | 0 (0) | 0 (0) | 38 (27) |”
Time-to-event outcome percentage uses an inconsistent denominator
MinorTable 3 covers 68 studies and lists 30 with time-to-event outcomes. That is 44%, not the reported 48%; the other outcome-type percentages use 68 as their denominator.
Reported 48 · recomputed 44.117647
“| Time to event | 48 (30) |”
Table 6 note mislabels the study-aim groups
MinorThe Table 6 note calls the groups of 48 and 17 predictor-finding and model-development studies. Elsewhere, those study types are counted as 51 and 14; 48 and 17 are the primary- and secondary-aim groups.
“48 predictor finding studies, 17 model development studies, or 65 total”
“| Type of Result Presented | Predictor Finding Studies (n = 51) | Development Studies (n = 14) | Total (n = 65)a |”
Change-in-C-statistic total does not equal subgroup counts
MinorTable 6 reports five primary-aim studies and zero secondary-aim studies using this criterion, but gives a total of six.
Reported 6 · recomputed 5
“| Change in C-statistic | 10; 14 (5) | 0; 0 (0) | 9; 13 (6) |”
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Self-citationAssessing the Generalizability of Prognostic Information
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Self-citationThe Risk of Determining Risk with Multivariable Models
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Prognosis and prognostic research: application and impact of prognostic models in clinical practice
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Self-citationInappropriate use of bivariable analysis to screen risk factors for use in multivariable analysis
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External validation is necessary in prediction research:
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Self-citationPredictive value of statistical models.
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Evidence of bias and variation in diagnostic accuracy studies
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Self-citationValidation, updating and impact of clinical prediction rules: A review
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Self-citationMethodologic Standards for the Development of Clinical Decision Rules in Emergency Medicine
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Self-citationCase–Control and Two-Gate Designs in Diagnostic Accuracy Studies
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Stepwise Selection in Small Data Sets A Simulation Study of Bias in Logistic Regression Analysis
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Theoretical Epidemiology: Principles of Occurrence Research in Medicine
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Updating methods improved the performance of a clinical prediction model in new patients
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Self-citationAdjusting for partially missing baseline measurements in randomized trials
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Systematic review of prognostic models in traumatic brain injury
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Clinical Epidemiology: Principles, Methods, And Applications For Clinical Research
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Methodological challenges in the evaluation of prognostic factors in breast cancer.
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Self-citationFive-Year Risk of Colorectal Neoplasia after Negative Screening Colonoscopy
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The use and reporting of multiple imputation in medical research – a review
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Burton, A., & Altman, D. G. (2004) · British Journal of Cancer · doi:10.1038/sj.bjc.6601907 · Cited by 219 · see citing works
Reporting methods in studies developing prognostic models in cancer: a review
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“Good reporting of how candidate predictors were pre-selected in our review compares favourably with other reviews [15,20,21,29,33,35,36,48].”
BackgroundResultSelf-citationPrehospital Termination of Resuscitation in Cases of Refractory Out-of-Hospital Cardiac Arrest
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Reporting performance of prognostic models in cancer: a review.
Mallett, S., Royston, P., Waters, R., Dutton, S., & Altman, D. G. (2010) · BMC Medicine · doi:10.1186/1741-7015-8-21 · Cited by 195 · see citing works
Self-citationKnol, M. J., Janssen, K. J. M., Donders, A. R. T., Egberts, T. C. G., Heerdink, E. R., Grobbee, D. E., Moons, K. G. M., & Geerlings, M. I. (2010) · Journal of Clinical Epidemiology · doi:10.1016/j.jclinepi.2009.08.028 · Cited by 190 · see citing works
Self-citationSample sizes of studies on diagnostic accuracy: literature survey.
Bachmann, L. M., Puhan, M. A., Riet, G. T., & Bossuyt, P. M. (2006) · BMJ · doi:10.1136/bmj.38793.637789.2f · Cited by 184 · see citing works
Mushkudiani, N. A., Hukkelhoven, C. W. P. M., Hernández, A. V., Murray, G. D., Choi, S. C., Maas, A. I. R., & Steyerberg, E. W. (2008) · Journal of Clinical Epidemiology · doi:10.1016/j.jclinepi.2007.06.011 · Cited by 177 · see citing works
Self-citationPrediction models in reproductive medicine: a critical appraisal†
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Manchester triage system in paediatric emergency care: prospective observational study
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Self-citationPrehypertension during Young Adulthood and Coronary Calcium Later in Life
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Marshall, A., Altman, D. G., & HOLDER, R. L. (2010) · BMC Medical Research Methodology · doi:10.1186/1471-2288-10-112 · Cited by 146 · see citing works
Self-citationHayden, J. A., Côté, P., Steenstra, I. A., & Bombardier, C. (2008) · Journal of Clinical Epidemiology · doi:10.1016/j.jclinepi.2007.08.005 · Cited by 138 · see citing works
Advantages of the nested case-control design in diagnostic research
Biesheuvel, C. J., Vergouwe, Y., Oudega, R., Hoes, A. W., Grobbee, D. E., & Moons, K. G. M. (2008) · BMC Medical Research Methodology · doi:10.1186/1471-2288-8-48 · Cited by 135 · see citing works
Self-citationPrognostic Models: A Methodological Framework and Review of Models for Breast Cancer
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Self-citationValidity of prognostic models: When is a model clinically useful?
Vergouwe, Y., Steyerberg, E. W., Eijkemans, M. J. C., & Habbema, J. D. F. (2002) · Seminars in Urologic Oncology · doi:10.1053/suro.2002.32521 · Cited by 123 · see citing works
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Self-citationComposite End Points in Randomized Trials: There Is No Free Lunch
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Composite outcomes in cardiovascular research: a survey of randomized trials.
Lim, E., Brown, A., Helmy, A., Mussa, S., & Altman, D. G. (2008) · Annals of Internal Medicine · doi:10.7326/0003-4819-149-9-200811040-00004 · Cited by 116 · see citing works
Self-citationBias arising from missing data in predictive models.
Gorelick, M. H. (2006) · Journal of Clinical Epidemiology · doi:10.1016/j.jclinepi.2004.11.029 · Cited by 107 · see citing works
Strengthening the Reporting of Genetic Risk Prediction Studies: The GRIPS Statement
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Primer: an evidence-based approach to prognostic markers
Altman, D. G., & Riley, R. D. (2005) · Nature Clinical Practice Oncology · doi:10.1038/ncponc0287 · Cited by 76 · see citing works
Self-citationStrengthening the reporting of Genetic RIsk Prediction Studies: the GRIPS Statement
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Self-citationCommentary: Epidemiology in the Right Direction: The Importance of Descriptive Research
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Prognostic Models: A Methodological Framework and Review of Models for Breast Cancer
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Self-citation- Self-citation
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QuPath: Open source software for digital pathology image analysis
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Self-citationCollins, G. S., Reitsma, J. B., Altman, D. G., & Moons, K. G. M. (2015) · BMJ · doi:10.1136/bmj.g7594 · Cited by 4,388 · see citing works
“Bouwmeester and colleagues (23) evaluated 71 reports, published in 2008 in 6 high-impact general medical journals, and likewise observed an overwhelmingly poor level of reporting.”
Self-citationCollins, G. S., Moons, K. G. M., Dhiman, P., Riley, R. D., Beam, A. L., Van Calster, B., Ghassemi, M., Liu, X., Reitsma, J. B., van Smeden, M., et al. (2024) · BMJ · doi:10.1136/bmj-2023-078378 · Cited by 4,718 · see citing works
Self-citationCollins, P. G. S., Reitsma, J. B., Altman, D. G., & Moons, K. G. M. (2015) · British Journal of Cancer · doi:10.1038/bjc.2014.639 · Cited by 3,312 · see citing works
“…have showed that the quality of reporting in published articles describing the development or validation of multivariable prediction models in medicine is poor (Laupacis et al, 1997; Ettema et al, 2010; Mallett et al, 2010; Collins et al, 2011; Bouwmeester et al, 2012; Collins et al, 2013, 2014).”
InfluentialSelf-citationCollins, G. S., Reitsma, J. B., Altman, D. G., & Moons, K. G. M. (2015) · Annals of Internal Medicine · doi:10.7326/m14-0697 · Cited by 3,267 · see citing works
“Bouwmeester and colleagues [23] evaluated 71 reports, published in 2008 in 6 high-impact general medical journals, and likewise observed an overwhelmingly poor level of reporting.”
BackgroundSelf-citationPROBAST: A Tool to Assess the Risk of Bias and Applicability of Prediction Model Studies
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“(1) Predictor finding studies (also known as risk factor or prognostic factor studies) aim to identify which predictors independently contribute to the prediction of a diagnostic or prognostic outcome.(1, 6) Prediction model studies typically aim to develop, validate or adjust (e.”
InfluentialSelf-citationCalibration: the Achilles heel of predictive analytics
On behalf of Topic Group ‘Evaluating diagnostic tests and prediction models’ of the STRATOS initiative, Van Calster, B., McLernon, D. J., van Smeden, M., Wynants, L., & Steyerberg, E. W. (2019) · BMC Medicine · doi:10.1186/s12916-019-1466-7 · Cited by 2,650 · see citing works
“Systematic reviews have found that calibration is assessed far less often than discrimination [2, 3, 5 – 7], which is problematic since poor calibration can make predictions misleading [8].”
BackgroundSelf-citationMoons, K. G. M., de Groot, J. A. H., Bouwmeester, W., Vergouwe, Y., Mallett, S., Altman, D. G., Reitsma, J. B., & Collins, G. S. (2014) · PLoS Medicine · doi:10.1371/journal.pmed.1001744 · Cited by 2,018 · see citing works
“Continuous or categorical predictors are frequently dichotomised for the analysis [2,42,56,60,78] despite strong evidence and recommendations to the contrary [92–94].”
BackgroundMethodInfluentialSelf-citationTowards better clinical prediction models: seven steps for development and an ABCD for validation
Steyerberg, E. W., & Vergouwe, Y. (2014) · European Heart Journal · doi:10.1093/eurheartj/ehu207 · Cited by 1,928 · see citing works
“least reported poorly (4) (5) (6) (7).”
Self-citationCollins, G. S., Reitsma, J. B., Altman, D. G., & Moons, K. (2015) · BMC Medicine · doi:10.1186/s12916-014-0241-z · Cited by 1,927 · see citing works
“These and other reviews provide a clear picture that, across different disease areas and different journals, there is a generally poor level of reporting of prediction model studies [6,2-27,29].”
InfluentialSelf-citationMoons, K. G. M., Wolff, R. F., Riley, R. D., Whiting, P. F., Westwood, M., Collins, G. S., Reitsma, J. B., Kleijnen, J., & Mallett, S. (2019) · Annals of Internal Medicine · doi:10.7326/m18-1377 · Cited by 1,669 · see citing works
Self-citationMa, L.-L., Wang, Y.-Y., Yang, Z.-H., Huang, D., Weng, H., & Zeng, X.-T. (2020) · Military Medical Research · doi:10.1186/s40779-020-00238-8 · Cited by 1,648 · see citing works
Reducing waste from incomplete or unusable reports of biomedical research
Glasziou, P., Altman, D. G., Bossuyt, P., Boutron, I., Clarke, M., Julious, S., Michie, S., Moher, D., & Wager, E. (2014) · The Lancet · doi:10.1016/s0140-6736(13)62228-x · Cited by 1,319 · see citing works
Self-citationNosek (2012) · doi:10.1177/1745691612459058 · Cited by 1,301 · see citing works
Leakage and the reproducibility crisis in machine-learning-based science
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Self-citationPrediction models for cardiovascular disease risk in the general population: systematic review
Damen, J. A. A. G., Hooft, L., Schuit, E., Debray, T. P. A., Collins, G. S., Tzoulaki, I., Lassale, C. M., Siontis, G. C. M., Chiocchia, V., Roberts, C., et al. (2016) · BMJ · doi:10.1136/bmj.i2416 · Cited by 935 · see citing works
“15 28 29-31 The full list of extracted items is available on request.”
Self-citationCollins, G. S., Reitsma, J. B., Altman, D. G., & Moons, K. G. M. (2015) · British Journal of Surgery · doi:10.1002/bjs.9736 · Cited by 889 · see citing works
Self-citationCollins, G. S., Dhiman, P., Andaur Navarro, C. L., Ma, J., Hooft, L., Reitsma, J. B., Logullo, P., Beam, A. L., Peng, L., Van Calster, B., et al. (2021) · BMJ Open · doi:10.1136/bmjopen-2020-048008 · Cited by 852 · see citing works
Self-citationSupervised Machine Learning: A Brief Primer
Jiang, T., Gradus, J. L., & Rosellini, A. J. (2020) · Behavior Therapy · doi:10.1016/j.beth.2020.05.002 · Cited by 880 · see citing works
“Finally, one underreported metric of model performance is calibration, or goodness of fit (Bouwmeester et al., 2012).”
BackgroundSample size for binary logistic prediction models: Beyond events per variable criteria
van Smeden, M., Moons, K. G. M., de Groot, J. A. H., Collins, P. G. S., Altman, D. G., Eijkemans, M. J. C., & Reitsma, J. B. (2018) · Statistical Methods in Medical Research · doi:10.1177/0962280218784726 · Cited by 818 · see citing works
“Binary logistic regression modeling is among the most frequently used approaches for developing multivariable clinical prediction models for binary outcomes.(1,2) Two major categories are: diagnostic prediction models that estimate the probability of a target disease being currently present versus not present; and pro…”
MethodSelf-citationCollins, G. S., Reitsma, J. B., Altman, D. G., & Moons, K. G. M. (2015) · Circulation · doi:10.1161/circulationaha.114.014508 · Cited by 793 · see citing works
“Bouwmeester and colleagues (23) evaluated 71 reports, published in 2008 in 6 high-impact general medical journals, and likewise observed an overwhelmingly poor level of reporting.”
Self-citationA guide to systematic review and meta-analysis of prognostic factor studies
Riley, R. D., Moons, K. G. M., Snell, K. I. E., Ensor, J., Hooft, L., Altman, D. G., Hayden, J. A., Collins, P. G. S., & Debray, T. P. A. (2019) · BMJ · doi:10.1136/bmj.k4597 · Cited by 754 · see citing works
Self-citationReporting of artificial intelligence prediction models
Collins, G. S., & Moons, K. G. M. (2019) · The Lancet · doi:10.1016/s0140-6736(19)30037-6 · Cited by 701 · see citing works
Self-citationCollins, G. S., de Groot, J. A., Dutton, S., Omar, O., Shanyinde, M., Tajar, A., Voysey, M., Wharton, R., Yu, L.-M., Moons, K. G., et al. (2014) · BMC Medical Research Methodology · doi:10.1186/1471-2288-14-40 · Cited by 729 · see citing works
“Both components are extremely important and should be reported for all studies evaluating the performance of a prediction model, yet calibration, which assesses how close the prediction for an individual is to their true risk, is inexplicably rarely reported, as observed in this and other reviews [1,23,47].”
BackgroundMethodResultInfluentialSelf-citationA guide to systematic review and meta-analysis of prediction model performance
Debray, T. P. A., Damen, J. A. A. G., Snell, K. I. E., Ensor, J., Hooft, L., Reitsma, J. B., Riley, R. D., & Moons, K. G. M. (2017) · BMJ · doi:10.1136/bmj.i6460 · Cited by 657 · see citing works
“12 27 Therefore, we propose to extract information on the total number of observed (O) and expected (E) events, which are statistics most likely to be reported or derivable (appendix 7).”
BackgroundSelf-citationRiley, R. D., Ensor, J., Snell, K. I. E., Debray, T. P. A., Altman, D. G., Moons, K. G. M., & Collins, G. S. (2016) · BMJ · doi:10.1136/bmj.i3140 · Cited by 585 · see citing works
“21 23 A major problem of external validation studies is that they are often based on small and local datasets.”
BackgroundSelf-citationDebray, T. P. A., Vergouwe, Y., Koffijberg, H., Nieboer, D., Steyerberg, E. W., & Moons, K. G. M. (2015) · Journal of Clinical Epidemiology · doi:10.1016/j.jclinepi.2014.06.018 · Cited by 597 · see citing works
“Differences in study-level characteristics (such as inclusion and exclusion criteria, details on subject recruitment, or study design choices) may provide additional insights into achieved model performance and could therefore further improve the interpretation of prediction model validation results (step 3) [47].”
Self-citationLogistic regression was as good as machine learning for predicting major chronic diseases
Nusinovici, S., Tham, Y. C., Yan, M. Y. C., Ting, D. S. W., Li, J., Sabanayagam, C., Wong, T. Y., & Cheng, C. (2020) · Journal of Clinical Epidemiology · doi:10.1016/j.jclinepi.2020.03.002 · Cited by 569 · see citing works
Siontis, G. C. M., Tzoulaki, I., Castaldi, P. J., & Ioannidis, J. P. A. (2014) · Journal of Clinical Epidemiology · doi:10.1016/j.jclinepi.2014.09.007 · Cited by 462 · see citing works
“Recently published systematic reviews of studies in prognostic research field have raised concerns about the proper validation and reporting strategies of new risk models [2,75,76].”
BackgroundCollins, G. S., Reitsma, J. B., Altman, D. G., & Moons, K. G. M. (2015) · European Urology · doi:10.1016/j.eururo.2014.11.025 · Cited by 439 · see citing works
Self-citationPavlou, M., Ambler, G., Seaman, S. R., Iorio, M. D., & Omar, R. (2015) · Statistics in Medicine · doi:10.1002/sim.6782 · Cited by 421 · see citing works
“[11], and is useful for quantifying the amount of information in the data relative to model complexity.”
BackgroundAndaur Navarro, C. L., Damen, J. A. A., Takada, T., Nijman, S. W. J., Dhiman, P., Ma, J., Collins, G. S., Bajpai, R., Riley, R. D., Moons, K. G. M., et al. (2021) · BMJ · doi:10.1136/bmj.n2281 · Cited by 415 · see citing works
“…publications have raised concern about the methodological quality of prediction models developed with conventional statistical techniques, 6 17 18 a formal methodological and risk of bias assessment of supervised machine learning based prediction model studies across all medical disciplines…”
BackgroundSelf-citationGerry, S., Bonnici, T., Birks, J., Kirtley, S., Virdee, P. S., Watkinson, P., & Collins, P. G. S. (2020) · BMJ · doi:10.1136/bmj.m1501 · Cited by 398 · see citing works
Calibration of Risk Prediction Models: Impact on Decision-Analytic Performance
Van Calster, B., & Vickers, A. J. (2015) · Medical Decision Making · doi:10.1177/0272989x14547233 · Cited by 371 · see citing works
Shaikhina, T., Lowe, D., Daga, S., Briggs, D., Higgins, R. M., & Khovanova, N. A. (2017) · Biomedical Signal Processing and Control · doi:10.1016/j.bspc.2017.01.012 · Cited by 363 · see citing works
Collins, P. G. S., Reitsma, J. B., Altman, D. G., & Moons, K. G. M. (2015) · Journal of Clinical Epidemiology · doi:10.1016/j.jclinepi.2014.11.010 · Cited by 333 · see citing works
“Bouwmeester and colleagues [23] evaluated 71 reports, published in 2008 in 6 high-impact general medical journals, and likewise observed an overwhelmingly poor level of reporting.”
Self-citationGuo, C., Ashrafian, H., Ghafur, S., Fontana, G., Gardner, C., & Prime, M. S. (2020) · npj Digital Medicine · doi:10.1038/s41746-020-00314-2 · Cited by 322 · see citing works
“Systematic reviews of prediction models are a new and evolving area and are increasingly undertaken to systematically identify, appraise, and summarize evidence on the performance of prediction models 71 – 73 .”
BackgroundCalibration drift in regression and machine learning models for acute kidney injury
Davis, S. E., Lasko, T. A., Chen, G., Siew, E. D., & Matheny, M. E. (2017) · Journal of the American Medical Informatics Association · doi:10.1093/jamia/ocx030 · Cited by 312 · see citing works
A framework for meta-analysis of prediction model studies with binary and time-to-event outcomes
Debray, T. P., Damen, J. A., Riley, R. D., Snell, K., Reitsma, J. B., Hooft, L., Collins, G. S., & Moons, K. G. (2019) · Statistical Methods in Medical Research · doi:10.1177/0962280218785504 · Cited by 278 · see citing works
“However, they are often poorly reported.(11,16) We now discuss how to retrieve the necessary but not explicitly reported predictive performance estimates from the primary prediction model (validation) studies using other reported data.”
BackgroundMethodSelf-citationCowley, L., Farewell, D. M., Maguire, S., & Kemp, A. (2019) · Diagnostic and Prognostic Research · doi:10.1186/s41512-019-0060-y · Cited by 265 · see citing works
Binuya, M. A. E., Engelhardt, E. G., Schats, W., Schmidt, M. K., & Steyerberg, E. W. (2022) · BMC Medical Research Methodology · doi:10.1186/s12874-022-01801-8 · Cited by 246 · see citing works
Self-citationDonzé, J. D., Williams, M. V., Robinson, E. J., Zimlichman, E., Aujesky, D., Vasilevskis, E. E., Kripalani, S. B., Metlay, J. P., Wallington, T., Fletcher, G. S., et al. (2016) · JAMA Internal Medicine · doi:10.1001/jamainternmed.2015.8462 · Cited by 241 · see citing works
Vistisen, D., Andersen, G. S., Hansen, C. S., Hulman, A., Henriksen, J. E., Bech-Nielsen, H., & Jørgensen, M. E. (2016) · Circulation · doi:10.1161/circulationaha.115.018844 · Cited by 229 · see citing works
“In addition, we used clinical judgment, as is recommended, to choose between highly correlated predictors of equal predictive power.(29) Before analysis, predictors with a highly skewed distribution (triglycerides, eGFR and TSH) were log-transformed to improve model calibration.”
BackgroundMethodInfluentialFundamentals of Clinical Data Science
Kubben, P., Dumontier, M., & Dekker, A. L. A. J. (2018) · Springer eBooks · doi:10.1007/978-3-319-99713-1 · Cited by 240 · see citing works
Guide to presenting clinical prediction models for use in clinical settings
Bonnett, L., Snell, K., Collins, G., & Riley, R. (2019) · British medical journal · doi:10.1136/bmj.l737 · Cited by 208 · see citing works
Diagnostic and prognostic prediction models
Hendriksen, J. M. T., Geersing, G. J., Moons, K. G. M., & de Groot, J. A. H. (2013) · Journal of Thrombosis and Haemostasis · doi:10.1111/jth.12262 · Cited by 229 · see citing works
Self-citationRisk Prediction Models for Patients With Chronic Kidney Disease
Tangri, N., Kitsios, G. D., Inker, L. A., Griffith, J. L., Naimark, D. M. J., Walker, S., Rigatto, C., Uhlig, K., Kent, D. M., & Levey, A. S. (2013) · Annals of Internal Medicine · doi:10.7326/0003-4819-158-8-201304160-00004 · Cited by 226 · see citing works
“Incorporating a prediction model into clinical practice requires an evaluation of its internal and external validity and clinical usefulness (10, 11).”
MethodEvaluating the impact of prediction models: lessons learned, challenges, and recommendations
Kappen, T. H., van Klei, W. A., van Wolfswinkel, L., Kalkman, C. J., Vergouwe, Y., & Moons, K. G. M. (2018) · Diagnostic and Prognostic Research · doi:10.1186/s41512-018-0033-6 · Cited by 215 · see citing works
“Currently, the number of published prediction models is overwhelming [3, 10, 55].”
BackgroundSelf-citationPrognostic models for breast cancer: a systematic review
Phung, M. T., Tin, S. T., & Elwood, M. (2019) · BMC Cancer · doi:10.1186/s12885-019-5442-6 · Cited by 214 · see citing works
A risk prediction score for acute kidney injury in the intensive care unit
Malhotra, R., KASHANI, K. B., Macedo, E. M., Kim, J., Bouchard, J., Wynn, S., Li, G., Lucila Ohno-machado, A., & Mehta, R. L. (2017) · Nephrology Dialysis Transplantation · doi:10.1093/ndt/gfx026 · Cited by 202 · see citing works
“Using the development cohort at UCSD, the regression coefficient-based models were constructed using a 5-fold crossvalidation procedure [60, 61].”
MethodIndividual Participant Data Meta-Analysis for a Binary Outcome: One-Stage or Two-Stage?
Debray, T. P. A., Moons, K. G. M., Abo-Zaid, G., Koffijberg, H., & Riley, R. D. (2013) · PLoS ONE · doi:10.1371/journal.pone.0060650 · Cited by 201 · see citing works
Self-citationKorolev, I. O., Symonds, L. L., Bozoki, A., & Initiative, A. D. N. (2016) · PLoS ONE · doi:10.1371/journal.pone.0138866 · Cited by 192 · see citing works
“As the next step, it will be important to externally validate our model on an independent dataset [18,59].”
BackgroundPrognostic models in obstetrics: available, but far from applicable
Kleinrouweler, C. E., Cheong-See, F., Collins, P. G. S., Kwee, A., Thangaratinam, S., Khan, K. S., Mol, B. W., Pajkrt, E., Moons, K. G. M., & Schuit, E. (2015) · American Journal of Obstetrics and Gynecology · doi:10.1016/j.ajog.2015.06.013 · Cited by 196 · see citing works
Self-citationWilimitis, D., & Walsh, C. G. (2023) · JMIR AI · doi:10.2196/49023 · Cited by 185 · see citing works
Collins, G. S., Ogundimu, E. O., Cook, J. A., Manach, Y. L., & Altman, D. G. (2016) · Statistics in Medicine · doi:10.1002/sim.6986 · Cited by 185 · see citing works
Self-citationA conceptual framework for prognostic research
Kent, P., Cancelliere, C., Boyle, E., Cassidy, J., & Kongsted, A. (2020) · BMC Medical Research Methodology · doi:10.1186/s12874-020-01050-7 · Cited by 138 · see citing works
Do prostate cancer risk models improve the predictive accuracy of PSA screening? A meta-analysis
Louie, K. S., Seigneurin, A., Cathcart, P. J., & Sasieni, P. D. (2014) · Annals of Oncology · doi:10.1093/annonc/mdu525 · Cited by 184 · see citing works
“[19] for methodological assessment of prediction research.”
Wang, S. V., Schneeweiß, S., Berger, M. L., Brown, J. S., de Vries, F., Douglas, I., Gagne, J. J., Gini, R., Klungel, O. H., Mullins, C. D., et al. (2017) · Pharmacoepidemiology and Drug Safety · doi:10.1002/pds.4295 · Cited by 182 · see citing works
“[16] Bouwmeester W, Zuithoff NP, Mallett S, et al.”
BackgroundPoor performance of clinical prediction models: the harm of commonly applied methods
Steyerberg, E. W., Uno, H., Ioannidis, J. P. A., CALSTER, B. V., Ukaegbu, C. I., Dhingra, T. G., Syngal, S., & Kastrinos, F. (2017) · Journal of Clinical Epidemiology · doi:10.1016/j.jclinepi.2017.11.013 · Cited by 166 · see citing works
Self-citationManuel, D. G., Perez, R., Sanmartin, C., Taljaard, M., Hennessy, D. A., Wilson, K. R., Tanuseputro, P., Manson, H. E., Bennett, C. L., Tuna, M., et al. (2016) · PLoS Medicine · doi:10.1371/journal.pmed.1002082 · Cited by 165 · see citing works
“Continuous measures were assessed and preferentially included for two reasons: for improved predictive performance (see below) [13,18,19], and to allow counterfactual examination of small changes in population exposure [12,13].”
Backgroundvan Loon, F. H. J., van Hooff, L. W. E., de Boer, H. D., Koopman, S. S. H. A., Buise, M. P., Korsten, H. H. M., Dierick-van Daele, A. T. M., & Bouwman, A. R. A. (2019) · Journal of Clinical Medicine · doi:10.3390/jcm8020144 · Cited by 157 · see citing works
“05 from the univariate analysis for examining their independent association with the primary outcome [14,18,20].”
MethodInfluentialA tutorial on methodological studies: the what, when, how and why
Mbuagbaw, L. C. E., Lawson, D. O., Puljak, L., Allison, D. B., & Thabane, L. (2020) · BMC Medical Research Methodology · doi:10.1186/s12874-020-01107-7 · Cited by 164 · see citing works
Peat, G., Riley, R. D., Croft, P. R., Morley, K. I., Kyzas, P., Moons, K. G. M., Perel, P., Steyerberg, E. W., Schroter, S., Altman, D. G., et al. (2014) · PLoS Medicine · doi:10.1371/journal.pmed.1001671 · Cited by 152 · see citing works
“Most do not explicitly build on previous findings, are often too small to answer the research question, and typically do not provide sample size calculations [24,25].”
BackgroundSelf-citationHeus, P., Damen, J. A., Pajouheshnia, R., Scholten, R. J. P. M., Reitsma, J. B., Collins, P. G. S., Altman, D. G., Moons, K. G. M., & Hooft, L. (2018) · BMC Medicine · doi:10.1186/s12916-018-1099-2 · Cited by 145 · see citing works
Self-citationBattle, C. E., Hutchings, H. A., Lovett, S., Bouamra, O., Jones, S., Sen, A., Gagg, J., Robinson, D., Hartford-Beynon, J., Williams, J. R., et al. (2014) · Critical Care · doi:10.1186/cc13873 · Cited by 144 · see citing works
“Discrimination was assessed with the c-statistic (equivalent to the area under the receiver operator curve) [17].”
BackgroundMethodInfluentialAidi, H. E., Adams, A. L. L., Moons, K. G. M., den Ruijter, H. M., Mali, W. P. T. M., Doevendans, P. A., Nagel, E. C., Schalla, S. M., Bots, M. L., & Leiner, T. (2014) · Journal of the American College of Cardiology · doi:10.1016/j.jacc.2013.11.048 · Cited by 141 · see citing works
“5 None C B B B C B B 18 (7–23) d CM, MI, CHF: 16 (14)”
MethodSelf-citationvan de Sande, D., Van Genderen, M. E., Smit, J. M., Huiskens, J., Visser, J. J., Veen, R. E. R., van Unen, E., BA, O. H., Gommers, D., & Bommel, J. V. (2022) · BMJ Health & Care Informatics · doi:10.1136/bmjhci-2021-100495 · Cited by 135 · see citing works
Screening for Chronic Obstructive Pulmonary Disease
Guirguis‐Blake, J., Senger, C. A., Webber, E. M., Mularski, R. A., & Whitlock, E. P. (2016) · JAMA · doi:10.1001/jama.2016.2654 · Cited by 138 · see citing works
“Shortness of breath, presence of phlegm or mucus, functional limitations due to breathing problems, smoking history, age Tsukuya et al,28 2015 Japan Smokers and nonsmokers ≥4 67 (60-74)a 73 (71-75)a 15 (12-17)a 97 (96-98)a 0.”
BackgroundMahar, A. L., Compton, C. C., Halabi, S., Hess, K. R., Weiser, M. R., & Groome, P. A. (2017) · Journal of Surgical Oncology · doi:10.1002/jso.24774 · Cited by 138 · see citing works
Dhiman, P., Ma, J., Andaur Navarro, C. L., Speich, B., Bullock, G., Damen, J. A. A., Hooft, L., Kirtley, S., Riley, R. D., Van Calster, B., et al. (2022) · BMC Medical Research Methodology · doi:10.1186/s12874-022-01577-x · Cited by 140 · see citing works
“However, compared to the number of prediction models that are developed, very few are used in clinical practice and many models contribute to research waste [14–17].”
BackgroundCollins, P. G. S., Reitsma, J. B., Altman, D. G., & Moons, K. G. M. (2015) · BJOG An International Journal of Obstetrics & Gynaecology · doi:10.1111/1471-0528.13244 · Cited by 141 · see citing works
Self-citationInfluence of Household Rat Infestation on Leptospira Transmission in the Urban Slum Environment
Costa, F., Ribeiro, G. S., Felzemburgh, R. D. M., Santos, N. D. J., Reis, R. B., dos Santos, A. C., Fraga, D. B. M., de Araújo, W. N., Santana, C., Childs, J. E., et al. (2014) · PLoS neglected tropical diseases · doi:10.1371/journal.pntd.0003338 · Cited by 136 · see citing works
“The use of mutually adjusted weights per predictor is the standard methodology to develop a prognostic risk score [30].”
MethodOnland, W., Debray, T. P. A., Laughon, M. M., Miedema, M., Cools, F., Askie, L. M., Asselin, J. M., Calvert, S., Courtney, S. E., Dani, C., et al. (2013) · BMC Pediatrics · doi:10.1186/1471-2431-13-207 · Cited by 133 · see citing works
“In contrast with the former mentioned quality assessment instruments, this instrument should address not only reporting issues like the STROBE, but assess all the different aspects of the development, validation and implementation of a prediction model, as described in a series of recently published articles [8-11,60].”
BackgroundMethodSelf-citationBlagus, R., & Lusa, L. (2015) · BMC Bioinformatics · doi:10.1186/s12859-015-0784-9 · Cited by 131 · see citing works
“Prediction models represent a valuable tool in the decision making process of clinicians and health policy makers and are extensively used in medicine [1], however the majority of prediction studies in high impact journals do not follow the current methodological recommendations, limiting their reliability and applica…”
BackgroundHenderson, J. T., Thompson, J. H., Burda, B. U., & Cantor, A. G. (2017) · JAMA · doi:10.1001/jama.2016.18315 · Cited by 131 · see citing works
Navarro, C. L. A., Damen, J. A., Takada, T., Nijman, S. W. J., Dhiman, P., Ma, J., Collins, P. G. S., Bajpai, R. C., Riley, R. D., Moons, K. G. M., et al. (2022) · BMC Medical Research Methodology · doi:10.1186/s12874-021-01469-6 · Cited by 136 · see citing works
“Previous systematic reviews have shown poor reporting of regression-based prediction model studies [7, 8, 10, 11].”
BackgroundSelf-citationMortality prediction models in the general trauma population: A systematic review
de Munter, L., Polinder, S., Lansink, K. W. W., Cnossen, M. C., Steyerberg, E. W., & de Jongh, M. A. C. (2016) · Injury · doi:10.1016/j.injury.2016.12.009 · Cited by 127 · see citing works
Self-citationAndaur Navarro, C. L., Damen, J. A. A., van Smeden, M., Takada, T., Nijman, S. W. J., Dhiman, P., Ma, J., Collins, G. S., Bajpai, R., Riley, R. D., et al. (2023) · Journal of Clinical Epidemiology · doi:10.1016/j.jclinepi.2022.11.015 · Cited by 121 · see citing works
Self-citationRahman, M. S., & Sultana, M. (2017) · BMC Medical Research Methodology · doi:10.1186/s12874-017-0313-9 · Cited by 120 · see citing works
“Although the choice of this cut-off has some criticisms [6] for not being based on scientific reasoning except empirical evidence, it is found useful for quantifying the amount of information in the data relative to model complexity [7, 8].”
BackgroundSnell, K. I. E., Archer, L., Ensor, J., Bonnett, L. J., Debray, T. P. A., Phillips, B., Collins, P. G. S., & Riley, R. D. (2021) · Journal of Clinical Epidemiology · doi:10.1016/j.jclinepi.2021.02.011 · Cited by 125 · see citing works
“Calibration and clinical utility, in particular, are often underappreciated [24-26].”
BackgroundCnossen, M. C., Winkler, E. A., Yue, J. K., Okonkwo, D. O., Valadka, A. B., Steyerberg, E. W., Lingsma, H. F., Manley, G. T., & the TRACK-TBI Investigators (2017) · Journal of Neurotrauma · doi:10.1089/neu.2016.4819 · Cited by 118 · see citing works
Self-citationEstimation of Newborn Risk for Child or Adolescent Obesity: Lessons from Longitudinal Birth Cohorts
Morandi, A., Meyre, D., Lobbens, S., KLEINMAN, K. P., Kaakinen, M., Rifas‐Shiman, S. L., Vatin, V., Gaget, S., Pouta, A. M., Hartikainen, A., et al. (2012) · PLoS ONE · doi:10.1371/journal.pone.0049919 · Cited by 117 · see citing works
“The main strengths include: the novelty and the potential strong public health impact of multivariate obesity predicting tools valid for newborns; the optimization of results reliability and robustness by the adoption of several recommended methods shown recently to be lacking in several recent high impact prediction…”
Machine Learning for Predictive Modelling based on Small Data in Biomedical Engineering
Shaikhina, T., Lowe, D., Daga, S., Briggs, D., Higgins, R. M., & Khovanova, N. A. (2015) · IFAC-PapersOnLine · doi:10.1016/j.ifacol.2015.10.185 · Cited by 115 · see citing works
Serghiou, S., Kyriakopoulou, A., & Ioannidis, J. P. A. (2016) · Molecular Cancer · doi:10.1186/s12943-016-0535-1 · Cited by 112 · see citing works
“dealing with prognosis [30, 38] and recommendations regarding reporting of biomarker studies [39, 40].”
BackgroundPrediction versus aetiology: common pitfalls and how to avoid them
van Diepen, M., Ramspek, C. L., Jager, K. J., Zoccali, C., & Dekker, F. W. (2017) · Nephrology Dialysis Transplantation · doi:10.1093/ndt/gfw459 · Cited by 113 · see citing works
Wang, S. V., Schneeweiß, S., Berger, M. L., Brown, J. S., de Vries, F., Douglas, I., Gagne, J. J., Gini, R., Klungel, O. H., Mullins, C. D., et al. (2017) · Value in Health · doi:10.1016/j.jval.2017.08.3018 · Cited by 108 · see citing works
“13-16 This has led to high profile controversies over apparent discrepancies in results and reduced confidence in evidence generated from healthcare databases.”
Dhiman, P., Ma, J., Navarro, C. L. A., Speich, B., Bullock, G. S., Damen, J. A., Kirtley, S., Hooft, L., Riley, R. D., CALSTER, B. V., et al. (2021) · Journal of Clinical Epidemiology · doi:10.1016/j.jclinepi.2021.06.024 · Cited by 109 · see citing works
“Results from several systematic reviews informally assessing reporting quality and TRIPOD adherence, in on-cology and otherwise, are comparable with our findings [ 6 , 8 , 22 , 24 , 25 , 36–38 ].”
BackgroundResultSelf-citationScientific Utopia: II. Restructuring incentives and practices to promote truth over publishabilityPreprint
Nosek, B. A., Spies, J. R., & Motyl, M. S. (2016) · doi:10.31219/osf.io/k4bgq · Cited by 114 · see citing works
Reuland, D. S., Brenner, A. T., Hoffman, R. M., McWilliams, A. D., Rhyne, R. L., Getrich, C. M., Tapp, H., Weaver, M. A., Callan, D., Cubillos, L., et al. (2017) · JAMA Internal Medicine · doi:10.1001/jamainternmed.2017.1294 · Cited by 110 · see citing works
Developing and validating risk prediction models in an individual participant data meta-analysis
Ahmed, I., Debray, T. P. A., Moons, K. G. M., & Riley, R. D. (2014) · BMC Medical Research Methodology · doi:10.1186/1471-2288-14-3 · Cited by 105 · see citing works
“In particular, our work allows us to provide some key recommendations for improving the design, conduct and reporting of future research this field (Table 3), which echo those for IPD meta-analysis of prognostic factor [37] and modelling [16] studies, and concur with previous [38,39] and ongoing [40] work toward impro…”
MethodSelf-citationGlucose homeostasis, obesity and diabetes
Bano, G. (2013) · Best Practice & Research Clinical Obstetrics & Gynaecology · doi:10.1016/j.bpobgyn.2013.02.007 · Cited by 104 · see citing works
Perel, P., Prieto‐Merino, D., Shakur‐Still, H., Clayton, T. C., Lecky, F. E., Bouamra, O., Russell, R. J., Faulkner, M., Steyerberg, E. W., & Roberts, I. (2012) · BMJ · doi:10.1136/bmj.e5166 · Cited by 103 · see citing works
Self-citationHodgson, L. E., Sarnowski, A., Roderick, P. J., Dimitrov, B. D., Venn, R. M., & Forni, L. G. (2017) · BMJ Open · doi:10.1136/bmjopen-2017-016591 · Cited by 100 · see citing works
Graafsma, J., Murphy, R. M., van de Garde, E. M. W., Karapinar‐Çarkit, F., Derijks, H. J., Hoge, R. H. L., Klopotowska, J. E., & van den Bemt, P. M. L. A. (2024) · Journal of the American Medical Informatics Association · doi:10.1093/jamia/ocae076 · Cited by 108 · see citing works
“53 Once the models are developed and validated, adequate attention must be given to their implementation into hospital practice.”
BackgroundFahey, M., Crayton, E., Wolfe, C. D. A., & Douiri, A. (2018) · PLoS ONE · doi:10.1371/journal.pone.0185402 · Cited by 96 · see citing works
Ma, G. K., & Ladabaum, U. (2014) · Clinical Gastroenterology and Hepatology · doi:10.1016/j.cgh.2014.01.042 · Cited by 97 · see citing works
“Although universally accepted criteria for study quality in the development of risk prediction models are not available, the investigators independently applied predefined criteria to assess themodel quality as good, fair, or poor on the basis of previously proposed principles.(36) Quality in studies reporting risk sc…”
MethodFredriksson, S. V., Alley, S., Rebar, A. L., Hayman, M., Vandelanotte, C., & Schöeppe, S. (2018) · PLoS ONE · doi:10.1371/journal.pone.0207003 · Cited by 96 · see citing works
- 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.
- ORCID
- Venue
- PLoS Medicine
- Publisher
- Public Library of Science (PLoS)
- Published
- 2012-05-22
- Volume
- 9
- Issue
- 5
- Pages
- e1001221
- ISSN
- 1549-1277 (ISSN-L)
- 1549-1676
- PMC
- PMC3358324
- OpenAlex
- W1998392635
- Semantic Scholar
- 2d86e73e56aa4394fe4ac9a6ed742d850abc73ce
- 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
- gold
- doi.org
- 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 › Health Professions › Health Information Management › 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
About these checks
Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.
Claims read by deepseek-v4.1-flash under definition version 3 (extract-v3) on 2026-10-09.
Classified by jev-1.13-20260917 (gold-guideline-v0.2) on 2026-10-09.
Statistics and arithmetic are recomputed, not judged.
Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.
What counts as a claim ›How we measure confidence ›How we check this ›
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