Targeted Genomic Region Masking Supports Accurate Variant Calling While Suppressing Low-Complexity Sequencing Artifacts

Background: False-positive variant calls generated within low-complexity regions (LCRs) remain a persistent bottleneck in clinical genomics, complicating downstream analysis. This study evaluates a targeted spatial masking strategy designed to suppress deterministic artifacts in short-read sequencing data, while preserving clinically actionable variants residing outside LCRs. We implemented a selective masking protocol prior to variant calling across analytical reference standards (EQA, NA12878) and two independent breast cancer whole-exome sequencing cohorts (n = 25). Methods: Callsets were evaluated for diagnostic sensitivity, precision gains, mutational signatures, VAF behavior, pseudo-multiallelic noise and ClinVar/dbSNP annotation. Results: The protocol removed thousands of sequencing and alignment artifacts while maintaining the retained biological callset, with negligible disease-associated diagnostic variants detected in the excluded artifact fraction. LCR masking preserved physiological Ti/Tv and Ins/Del profiles in retained calls, resolved pseudo-multiallelic noise, and distinguished excluded artifact calls by distorted mutational and VAF signatures. dbSNP profiling showed cohort-dependent behavior: TCGA-BRCA reproduced an intriguing phenomenon, with excluded calls showing higher dbSNP annotation than retained calls, whereas AURORA showed the opposite direction. Conclusions: These findings demonstrate the potential vulnerability of one-dimensional database annotation for variant authentication and highlight targeted spatial filtration as a critical, early pipeline intervention for high-fidelity clinical genomics of non-LCR-associated germline variants using short reads.

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Publication Details

Journal
Genes
Published
2026-06-30
DOI
https://doi.org/10.3390/genes17070772
Primary Topic
Genomics and Rare Diseases
Type
article
Field-Weighted Citation Impact
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article

Targeted Genomic Region Masking Supports Accurate Variant Calling While Suppressing Low-Complexity Sequencing Artifacts

Timokratis Karamitros, Vasiliki Pogka, Dimitrios Christos Tremoulis, Athina Tsagkalidou et al.
Genes
Genomics and Rare Diseases
article

Targeted Genomic Region Masking Supports Accurate Variant Calling While Suppressing Low-Complexity Sequencing Artifacts

Timokratis Karamitros, Vasiliki Pogka, Dimitrios Christos Tremoulis, Athina Tsagkalidou, Chrysoula Kaligerou
article en

Abstract

Background: False-positive variant calls generated within low-complexity regions (LCRs) remain a persistent bottleneck in clinical genomics, complicating downstream analysis. This study evaluates a targeted spatial masking strategy designed to suppress deterministic artifacts in short-read sequencing data, while preserving clinically actionable variants residing outside LCRs. We implemented a selective masking protocol prior to variant calling across analytical reference standards (EQA, NA12878) and two independent breast cancer whole-exome sequencing cohorts (n = 25). Methods: Callsets were evaluated for diagnostic sensitivity, precision gains, mutational signatures, VAF behavior, pseudo-multiallelic noise and ClinVar/dbSNP annotation. Results: The protocol removed thousands of sequencing and alignment artifacts while maintaining the retained biological callset, with negligible disease-associated diagnostic variants detected in the excluded artifact fraction. LCR masking preserved physiological Ti/Tv and Ins/Del profiles in retained calls, resolved pseudo-multiallelic noise, and distinguished excluded artifact calls by distorted mutational and VAF signatures. dbSNP profiling showed cohort-dependent behavior: TCGA-BRCA reproduced an intriguing phenomenon, with excluded calls showing higher dbSNP annotation than retained calls, whereas AURORA showed the opposite direction. Conclusions: These findings demonstrate the potential vulnerability of one-dimensional database annotation for variant authentication and highlight targeted spatial filtration as a critical, early pipeline intervention for high-fidelity clinical genomics of non-LCR-associated germline variants using short reads.

GenesVol. 17(7)
Pasteur Hellenic Institute (GR)
Good health and well-being
Openalex Percentile: Top 7%
Genomics and Rare Diseases
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