VAE-MS: An Asymmetric Variational Autoencoder for Mutational Signature Extraction
Authors: Ida Egendal, Rasmus Froberg Brøndum, Dan J Woodcock, Christopher Yau, Martin Bøgsted
Organizations: Center for Clinical Data Science, Aalborg University and Aalborg University Hospital, Aalborg, 9260, Denmark. · Clinical Cancer Research Center, Aalborg University Hospital, Aalborg, 9000, Denmark. · Nuffield Department of Surgical Sciences, University of Oxford, Oxford, OX3 9DU, England. · Nuffield Department of Women’s and Reproductive Health, University of Oxford, Oxford, OX3 9DU, England.
Mutational signature analysis has emerged as a powerful method for uncovering the underlying biological processes driving cancer development. However, the signature extraction process, typically performed using non-negative matrix factorization (NMF), often lacks reliability and clinical applicability. To address these limitations, several solutions have been introduced, including the use of neural networks to achieve more accurate estimates and probabilistic methods to better capture natural variation in the data. In this work, we introduce a Variational Autoencoder for Mutational Signatures (VAE-MS), a novel model that leverages both an asymmetric architecture and probabilistic methods for the extraction of mutational signatures. VAE-MS is compared to with three state-of-the-art models for mutational signature extraction: SigProfilerExtractor, the NMF-based gold standard; MUSE-XAE, an autoencoder that employs an asymmetric design without probabilistic components; and SigneR, a Bayesian NMF model, to illustrate the strength in combining a nonlinear extraction with a probabilistic model. In the ability to reconstruct input data and generalize to unseen data, models with probabilistic components (VAE-MS, SigneR) dramatically outperformed models without (SigProfilerExtractor, MUSE-XAE). The NMF-baed models (SigneR, SigProfilerExtractor) had the most accurate reconstructions in simulated data, while VAE-MS reconstructed more accurately on real cancer data. Upon evaluating the ability to extract signatures consistently, no model exhibited a clear advantage over the others. Software for VAE-MS is available at https://github.com/CLINDA-AAU/VAE-MS.
Figures & tables
Figure 1: Schematic representation of VAE-MS. BN: batch normalization.
Figure 2: Beeswarm plots of the number of signatures suggested by each model for extraction in S8 (A), S14 (B), and PCAWG (C) over the 10 splits by all compared models. The true number of signatures in the simulated datasets is highlighted with a tan line.
DKL
DMSE
Signature
Exposures in CI
Data
Model
Train
Test
Train
Test
ACS
PACS
SS
Train
Test
S8
VAE-MS
13.1
5.9
407
421
0.96
0.99
0.98
8.4%
7.3%
SigneR
7.9
3.1
2
5
0.99
0.99
0.99
36%
24%
SigProEx
3.1
3.2
0.12
12
0.99
0.99
0.99
-
-
MUSE-XAE
4.2
4.2
458
465
0.99
0.99
0.99
-
-
S14
VAE-MS
9.1
5.2
2231
2347
0.80
0.99
0.48
24.4%
24.6%
Table 1: The training and test performance of all models on the simulated data sets S8 and S14 with the number of signatures fixed at the true number of signatures. DKL : Kullback-Leibler divergence, DMSE : Mean-square error, ACS: Average Cosine Similarity to true signature set, PACS: Pairwise Average Cosine Similarity, SS: Silhouette Score, SigProEx: SigProfilerExtractor. Lowest error ( DKL,DMSE ), highest similarity measures (ACS, PACS, and SS) and highest fraction of exposures contained in CI are highlighted with bold text .
Figure 3: 2D t-SNE embeddings of the estimated signatures across the 10 splits of the simulated data sets, with the number of signatures fixed to the true value. Embeddings of the true signatures are indicated with black ’x’ markers.
Figure 4: Density plots of the true and estimated exposures for the corresponding signature for the second split of the S8 data.
DKL
DMSE
Model
Train
Test
Train
Test
PACS
S8
VAE-MS
11.7
4.9
255
255
0.99
SigneR
7.9
3.1
2
5
0.99
SigProfilerExtractor
3.1
3.2
0.12
12
0.99
MUSE-XAE
3.1
3.1
3.6
1.6
0.99
S14
VAE-MS
4.7
4.5
1817
1861
0.96
Table 2: Training and test performance of all models on the simulated and PCAWG data with the number of signatures selected by the models. DKL : Kullback-Leibler divergence, DMSE : Mean-squared error, PACS: Pairwise Average Cosine Similarity. Lowest error ( DKL,DMSE ), highest PACS are highlighted with bold text .
Variational autoencoders (VAEs) trained on multiple sequence alignments (MSAs) have emerged as powerful generative models for biological sequences, with applications ranging from disease variant prediction to functional RNA design. However, standard biological VAE training treats all sequences as exchangeable, ignoring the rich evolutionary structure that organizes homologous sequences from evolutionarily close to highly divergent. We propose Evolutionary Curriculum Learning (ECL), a training strategy that exploits this structure by progressively exposing the model to sequences of increasing evolutionary distance from sampled anchors, following a power-law expansion schedule. Applied to two architecturally distinct VAE models and two biological domains--protein variant effect prediction with EVE and RNA family sequence generation with RfamGen--ECL improves downstream task performance across five random seeds per configuration. Mean ClinVar classification AUROC rises from 0.981 to 0.989 for p53; for PTEN, ECL attains 1.000 in every seed whereas the baseline is unstable (mean 0.905, falling as low as 0.54). For RNA, ECL raises mean covariance-model bit scores on all three families tested and exceeds its seed-matched baseline in 12 of 15 training runs, though with only three families the effect cannot be established as significant at the family level. Ablation experiments show that progressively expanding the sampled sequences by evolutionary distance outperforms fixed-size neighborhood sampling in addition to uniform random sampling. Evolutionary distance is therefore a useful inductive bias for ordering the training curriculum in biological sequence modeling.
Richard Zhu, Kento Nishi
Department of Computer Science, Harvard University, Cambridge, MA, USA · Department of Statistics, Harvard University, Cambridge, MA, USA.
Longitudinal tumor measurements, dropout information, and genetic covariates provide complementary information about treatment response, but integrating these data sources within a single population modeling framework remains challenging. We extend the empirical Bayes variational autoencoder (EB-VAE) framework to joint longitudinal and time-to-event modeling and evaluate it on tumor growth data. The framework represents inter-individual variability using latent individual effects regularized by a covariate-conditioned empirical Bayes prior, while a decoder maps these latent effects to tumor-volume trajectories. To account for informative dropout, the decoder was augmented with a hazard model, yielding joint predictions of tumor growth and time to dropout. We further compared fully neural and hybrid semi-mechanistic decoder formulations and incorporated genomic covariates through a genetics-conditioned prior adaptation. The hybrid decoder recovered treatment-effect parameters broadly consistent with previously reported nonlinear mixed-effects estimates, while achieving prior predictive performance comparable to the neural decoder. The joint model reproduced both tumor-volume distributions and dropout patterns in held-out individuals, and genetic conditioning improved individual-level prior predictions in both cutaneous melanoma and breast cancer experiments. Stability selection identified several biologically plausible genetic indicators, including alterations in BRAF, NRAS, NF1, and MDM2. These results demonstrate that EB-VAE provides a flexible probabilistic framework for combining neural dynamics, mechanistic structure, time-to-event modeling, and high-dimensional covariates in pharmacometric applications.
Anders Sjöberg, Nils Olsson, Marcus Baaz +1
Fraunhofer-Chalmers Centre, Gothenburg, SE-412 88, Sweden · Department of Electrical Engineering, Chalmers University of Technology, Gothenburg, SE-412 96, Sweden
Multimodal medical data captures complementary aspects of disease. It provides a comprehensive basis for diagnosis and the identification of biological associations linked to progression or treatment response. Established contrastive learning approaches exploit shared information across modalities, but fall short in exploiting complementary, modality-specific signals relevant for a task. Here, we propose a synergistic multimodal variational autoencoder (VAE) framework that separates task-relevant unimodal and multimodal information from instance-specific variation task-irrelevant, allowing complementary signals across modalities to be leveraged for prediction. The method yields benefits even if modalities are missing at test time. We evaluate the method on a treatment response prediction task using medical imaging and RNA data. Empirical comparison with alignment-based and multimodal VAE state-of-the-art approaches demonstrates improved multimodal prediction performance, and consistent benefits for prediction from a single modality after multimodal training. The full code can be found here: https://github.com/cirmuw/SYNPRED.
Ivana Janíčková, Yen Y. Tan, Thomas H. Helbich +6
Computational Imaging Research Lab, Department of Biomedical Imaging and Image-guided Therapy, Medical University of Vienna, Austria · Comprehensive Center for Artificial Intelligence in Medicine, Medical University of Vienna, Austria · Christian Doppler Laboratory for Machine Learning Driven Precision Imaging, Department of Biomedical Imaging and Image-guided Therapy, Medical University of Vienna, Austria +3