Chris Humphries, Alastair M. Kilpatrick, Kathleen M. Scullion, Rhona Aird, Lorraine Bruce, Maria Elena Candela, Tak Yung Man, Stuart J. Forbes, James W. Dear
Clinical translation of novel therapies can be hindered by heterogeneity-driven sample size inflation in late-stage trials. In acetaminophen-induced liver injury (APAP DILI), many patients recover spontaneously, diluting investigational drug efficacy signals. We developed a prognostic enrichment tool to identify patients with worsening injury trajectories for more efficient trial designs. Biomarker model discovery and evaluation used serum samples from three UK cohorts: the MAPP2 APAP DILI biobank (n = 147), an independent pre-intervention evaluation cohort from the ongoing MAIL trial (n = 34), and healthy controls (n = 13). We measured 63 biomarkers and evaluated 321,682 combinations using kernel naïve Bayes classification to predict liver injury trajectory (ALT rising vs. falling). Sensitivity analysis using patient-level grouped cross-validation showed combining multiple biomarkers while constraining collinearity was necessary to maximize performance. A four-biomarker model (MCSFR, WBC, Sodium, K18) achieved AUC 0.868 (derivation) and 0.854 (evaluation). When optimized for prognostic certainty, the model yielded a Positive Likelihood Ratio of 14.4, increasing the Positive Predictive Value for worsening injury from a baseline of 29.4% to 85.7%. Time-dependent cost-minimization modeling for a hypothetical phase 3 trial identified an application threshold (sensitivity 80.0%, specificity 91.7%, Number Needed to Screen 3.4) as the global economic optimum, resulting in an illustrative trial cost reduction from $39.0 M to $8.3 M. This proof-of-concept demonstrates multidimensional biomarker models can resolve signal dilution. Distinguishing patients destined for injury progression reduces sample size requirements, which could de-risk novel therapy development.