Development and Internal Validation of a Multivariable Prediction Model for Adrenocortical-Carcinoma-Specific Mortality

Cancers, vol. 12, pp. 2720

Abstract

Adrenocortical carcinoma (ACC) has an incidence of about 1.0 per million per year. In general, survival of patients with ACC is limited. Predicting survival outcome at time of diagnosis is a clinical challenge. The aim of this study was to develop and internally validate a clinical prediction model for ACC-specific mortality. Data for this retrospective cohort study were obtained from the nine centers of the Dutch Adrenal Network (DAN). Patients who presented with ACC between 1 January 2004 and 31 October 2013 were included. We used multivariable Cox proportional hazards regression to compute the coefficients for the prediction model. Backward stepwise elimination was performed to derive a more parsimonious model. The performance of the initial prediction model was quantified by measures of model fit, discriminative ability, and calibration. We undertook an internal validation step to counteract the possible overfitting of our model. A total of 160 patients were included in the cohort. The median survival time was 35 months, and interquartile range (IQR) 50.7 months. The multivariable modeling yielded a prediction model that included age, modified European Network for the Study of Adrenal Tumors (mENSAT) stage, and radical resection. The c-statistic was 0.77 (95% Confidence Interval: 0.72, 0.81), indicating good predictive performance. We developed a clinical prediction model for ACC-specific mortality. ACC mortality can be estimated using a relatively simple clinical prediction model with good discriminative ability and calibration.

Authors 13

1. Madeleine H. T. Ettaieb corresponding

   Máxima Medisch Centrum

   Affiliation as printed

   Department of Internal Medicine, Division of Endocrinology, Máxima Medical Center, 5631 Eindhoven/Veldhoven, The Netherlands

2. Sander M. J. van Kuijk

   Maastricht University Medical Centre · Maastricht University

   Affiliation as printed

   Department of Clinical Epidemiology and Medical Technology Assessment, Maastricht University Medical Center, 6229 Maastricht, The Netherlands

3. Annelies de Wit-Pastoors

   Máxima Medisch Centrum

   Affiliation as printed

   Department of Internal Medicine, Division of Endocrinology, Máxima Medical Center, 5631 Eindhoven/Veldhoven, The Netherlands

4. Richard Abraham Feelders

   Erasmus MC

   Affiliation as printed

   Department of Internal Medicine, Division of Endocrinology, Erasmus Medical Center, 3015 Rotterdam, The Netherlands

5. Eleonora P.M. Corssmit Aachen Department of Internal Medicine Division of Endocrinology, Leiden University Medical Center

   Leiden University · Leiden University Medical Center

   Affiliation as printed

   Department of Internal Medicine, Division of Endocrinology, Leiden University Medical Center, 2333 Leiden, The Netherlands

6. Elisabeth M. W. Eekhoff

   Amsterdam UMC Location Vrije Universiteit Amsterdam

   Affiliation as printed

   Department of Internal Medicine, Division of Endocrinology, VU Medical Center, 1081 Amsterdam, The Netherlands

7. Paul van der Valk

   Amsterdam University Medical Centers · Amsterdam UMC Location Vrije Universiteit Amsterdam

   Affiliation as printed

   Department of Pathology, Location VU Medical Center, Amsterdam University Medical Centers, 1081 Amsterdam, The Netherlands

8. Henri J L M Timmers

   Radboud University Nijmegen · Radboud University Medical Center

   Affiliation as printed

   Department of Internal Medicine, Division of Endocrinology, Radboud University Medical Center, 6525 Nijmegen, The Netherlands

9. Michiel N. Kerstens

   University Medical Center Groningen · University of Groningen

   Affiliation as printed

   Department of Endocrinology, University of Groningen, University Medical Center Groningen, 9713 Groningen, The Netherlands

10. Heinz‐Josef Klümpen

    Amsterdam University Medical Centers · Cancer Center Amsterdam · University of Amsterdam

    Affiliation as printed

    Department of Medical Oncology, Amsterdam University Medical Centers, Cancer Center Amsterdam, University of Amsterdam, 1105 Amsterdam, The Netherlands

11. Rachel S. van Leeuwaarde

    University Medical Center Utrecht

    Affiliation as printed

    Department of Endocrine Oncology, University Medical Center Utrecht, 3584 Utrecht, The Netherlands

12. Bas Havekes

    Maastricht University Medical Centre · Maastricht University

    Affiliation as printed

    Department of Internal Medicine, Division of Endocrinology and Metabolism, Maastricht University Medical Center, 6229 Maastricht, The Netherlands

13. Harm Reinout Haak

    Maastricht University Medical Centre · Maastricht University · Máxima Medisch Centrum · Care and Public Health Research Institute

    Affiliation as printed

    CAPHRI School for Public Health and Primary Care, Ageing and Long-Term Care, 6229 Maastricht, The Netherlands

    Department of Internal Medicine, Division of Endocrinology, Máxima Medical Center, 5631 Eindhoven/Veldhoven, The Netherlands

    Department of Internal Medicine, Division of General Internal Medicine, Maastricht University Medical Center, 6229 Maastricht, The Netherlands