Molecular Diagnosis as a Probability of Necessity
Belmont, J. W.; Williams, C. J.; Shaw, C. A.
Show abstract
Genetic diagnosis requires attributing a patient's disease to a variant. We develop the Probability of Necessity (PN), a counterfactual estimand giving the probability that disease would not have occurred absent a germline variant, given both are observed; and we use this formalism to explore the logic and limitations of attributing a disease to genetic variants. We show PN dissociates from penetrance: identical penetrance yields different PN depending on baseline disease prevalence. Case selection biases PN estimation, favoring population-scale cohorts over ascertained case series. PN falls as competing-cause prevalence rises, formalizing why necessity differs from penetrance under causal heterogeneity. We introduce the Probability of Mediated Necessity (PMN), updating PN using mediator biomarkers proxying a variant's operative pathway. Using UK Biobank data, we estimate PN and PMN for rare LDLR variants in ischemic heart disease (IHD, ICD-10 I25), using hyperlipidemia (E78) as mediator, stratifying by ClinVar class and computational pathogenicity score. PN resolves heterogeneity within the variant of unknown significance (VUS) class beyond classification alone. Replicating in GBA1 carriers with Parkinson's disease, a mechanistically distinct, low-penetrance system, reproduces this ordering and shows high PN despite modest penetrance. These results support further development of causal necessity as a framework in genetic diagnostics with implications for policy and clinical decisions in both monogenic and common complex diseases.
Matching journals
The top 3 journals account for 50% of the predicted probability mass.
Similar papers in this journal
- The Causal Pivot: A Structural Approach to Genetic Heterogeneity and Variant Discovery in Complex Diseases 95%
- Estimating disease heritability from complex pedigrees allowing for ascertainment and covariates 94%
- The relationship between genotype- and phenotype-based estimates of genetic liability to psychiatric disorders, in practice and in theory. 94%
Similar papers in this journal
- Discovering genetic interactions bridging pathways in genome-wide association studies 94%
- Sharing information between related diseases using Bayesian joint fine mapping increases accuracy and identifies novel associations in six immune mediated diseases 94%
- MutPred2: inferring the molecular and phenotypic impact of amino acid variants 94%
Similar papers in this journal
- Continuous Bayesian Variant Interpretation Accounts for Incomplete Penetrance among Mendelian Cardiac Channelopathies 93%
- Disease-specific variant pathogenicity prediction significantly improves variant interpretation in inherited cardiac conditions 92%
- Evidence Aggregator: AI reasoning applied to rare disease diagnostics 91%
Similar papers in this journal
"Similar papers" are the closest papers from that journal in the model's embedding space. They show what the match is built on, but the ranking comes mostly from a classifier over the whole training set, not from these examples alone.