Back

Enhancing Missense Variant Classification in Predicted Intrinsically Disordered Regions

Gnanaolivu, R.; Hart, S.

2025-08-12 bioinformatics
10.1101/2025.08.08.669269 bioRxiv
Show abstract

The accurate classification of missense variants is a fundamental challenge in genomics, particularly for those within intrinsically disordered regions (IDRs) where the performance of existing computational predictors is suboptimal. To address this, we developed a machine learning model that extends traditional missense tools with properties that infer globular IDR conformation, phase separation, and protein embeddings. Using ClinVar variant classifications as ground truth, AlphaMissense, EVE, and ESM1b were the highest scoring unsupervised in silico missense predictors for IDR variants. Our baseline model, using only IDR-specific features achieved competitive performance on the hold-out test set with a PR-AUC of 0.800. Critically, when these IDR features were combined with these methods we saw significant overall improvement. The AlphaMissense-Enhanced model increased its PR-AUC from 0.807 to 0.931. Similarly, ESM1b-Enhanced improved PR-AUC from 0.679 to 0.878 and EVE increased from 0.591 to 0.918. These results demonstrate the effectiveness of our enhancements for classifying missense variants in IDRs and highlight its ability to complement existing in silico missense predictors.

Published in PLOS One · not in our set (fewer than 10 published preprints to learn from) · training set

Matching journals

The top 8 journals account for 50% of the predicted probability mass.

50% of probability mass above

"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.