Gene pathogenicity prediction of Mendelian diseases via the Random Forest algorithm
Sijie HeiD, Weiwei CheniD, Hankui LiuiD, Shengting LiiD, Dongzhu LeiiD, Xiao DangiD, Yulan CheniD, Xiuqing ZhangiD, Jianguo ZhangiD
Abstract
The study of Mendelian diseases and the identification of their causative genes are of great significance in the field of genetics. The evaluation of the pathogenicity of genes and the total number of Mendelian disease genes are both important questions worth studying. However, very few studies have addressed these issues to date, so we attempt to answer them in this study. We calculated gene pathogenicity prediction (GPP) score by a machine learning approach (random forest algorithm) to evaluate the pathogenicity of genes. When we applied the GPP score to the testing gene set, we obtained accuracy of 80%, recall of 93% and area under the curve (AUC) of 0.87. Our results estimated that a total of 10,399 protein-coding genes were Mendelian disease genes. Furthermore, we found the GPP score was positively correlated with the severity of disease. Our results indicate that GPP score may provide a robust and reliable guideline to predict the pathogenicity of protein-coding genes. To our knowledge, this is the first trial to estimate the total number of Mendelian disease genes.
02 /Specification & Classification
Click any tag to browse matching papers- Citations
- 7
- FWCI
- 0.75
- Field percentile
- top 31%
- Research Focus
- Approach
Reproducibility ledger
Nobody has reported trying to reproduce this paper yet. Did you replicate an experiment or run the code? Your report saves the next lab a week.
Sign in to file a reproducibility report.
Related papers
- MENDELSEEK: An algorithm that predicts Mendelian Genes and elucidates what makes them special
Hongyi Zhou, Brice Edelman, Jeffrey Skolnick·PLoS Computational BiologyPeer-reviewed·Feb 17, 2026
- G2P: Using machine learning to understand and predict genes causing rare neurological disorders
Juan Antonio Botía, Sebastian Guelfi, … Mina Ryten·bioRxiv·Mar 27, 2018
- DEVELOPING GENE-SPECIFIC META-PREDICTOR OF VARIANT PATHOGENICITY
Anna Rychkova, MyMy C. Buu, … Carlos D. Bustamante·bioRxiv·Mar 10, 2017
- Machine learning model to predict risk assessment of a child inheriting a genetic disorder
Medha Ramaswamy, Shreya Senthilnathan, … Karthikeyan Sivashanmugam·bioRxiv·Aug 22, 2024
- MendelVar: gene prioritization at GWAS loci using phenotypic enrichment of Mendelian disease genes
Maria Sobczyk, Tom R. Gaunt, Lavinia Paternoster·BioinformaticsPeer-reviewed·Jan 1, 2021
- Prevalence of loss-of-function, gain-of-function and dominant-negative mechanisms across genetic disease phenotypes
Mihaly Badonyi, Joseph A. Marsh·Nature CommunicationsPeer-reviewed·Sep 25, 2025