Differential methylation of circulating free DNA assessed through cfMeDiP as a new tool for breast cancer diagnosis and detection of BRCA1/2 mutation

Abstract Background Recent studies have highlighted the importance of the cell-free DNA (cfDNA) methylation profile in detecting breast cancer (BC) and its different subtypes. We investigated whether plasma cfDNA methylation, using cell-free Methylated DNA Immunoprecipitation and High-Throughput Seq...

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Main Authors: Piera Grisolia, Rossella Tufano, Clara Iannarone, Antonio De Falco, Francesca Carlino, Cinzia Graziano, Raffaele Addeo, Marianna Scrima, Francesco Caraglia, Anna Ceccarelli, Pier Vitale Nuzzo, Alessia Maria Cossu, Stefano Forte, Raffaella Giuffrida, Michele Orditura, Michele Caraglia, Michele Ceccarelli
Format: Article
Language:English
Published: BMC 2024-10-01
Series:Journal of Translational Medicine
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Online Access:https://doi.org/10.1186/s12967-024-05734-2
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author Piera Grisolia
Rossella Tufano
Clara Iannarone
Antonio De Falco
Francesca Carlino
Cinzia Graziano
Raffaele Addeo
Marianna Scrima
Francesco Caraglia
Anna Ceccarelli
Pier Vitale Nuzzo
Alessia Maria Cossu
Stefano Forte
Raffaella Giuffrida
Michele Orditura
Michele Caraglia
Michele Ceccarelli
author_facet Piera Grisolia
Rossella Tufano
Clara Iannarone
Antonio De Falco
Francesca Carlino
Cinzia Graziano
Raffaele Addeo
Marianna Scrima
Francesco Caraglia
Anna Ceccarelli
Pier Vitale Nuzzo
Alessia Maria Cossu
Stefano Forte
Raffaella Giuffrida
Michele Orditura
Michele Caraglia
Michele Ceccarelli
author_sort Piera Grisolia
collection DOAJ
description Abstract Background Recent studies have highlighted the importance of the cell-free DNA (cfDNA) methylation profile in detecting breast cancer (BC) and its different subtypes. We investigated whether plasma cfDNA methylation, using cell-free Methylated DNA Immunoprecipitation and High-Throughput Sequencing (cfMeDIP-seq), may be informative in characterizing breast cancer in patients with BRCA1/2 germline mutations for early cancer detection and response to therapy. Methods We enrolled 23 BC patients with germline mutation of BRCA1 and BRCA2 genes, 19 healthy controls without BRCA1/2 mutation, and two healthy individuals who carried BRCA1/2 mutations. Blood samples were collected for all study subjects at the diagnosis, and plasma was isolated by centrifugation. Cell-free DNA was extracted from 1 mL of plasma, and cfMeDIP-seq was performed for each sample. Shallow whole genome sequencing was performed on the immuno-precipitated samples. Then, the differentially methylated 300-bp regions (DMRs) between 25 BRCA germline mutation carriers and 19 non-carriers were identified. DMRs were compared with tumor-specific regions from public datasets to perform an unbiased analysis. Finally, two statistical classifiers were trained based on the GLMnet and random forest model to evaluate if the identified DMRs could discriminate BRCA-positive from healthy samples. Results We identified 7,095 hypermethylated and 212 hypomethylated regions in 25 BRCA germline mutation carriers compared to 19 controls. These regions discriminate tumors from healthy samples with high accuracy and sensitivity. We show that the circulating tumor DNA of BRCA1/2 mutant breast cancers is characterized by the hypomethylation of genes involved in DNA repair and cell cycle. We uncovered the TFs associated with these DRMs and identified that proteins of the Erythroblast Transformation Specific (ETS) family are particularly active in the hypermethylated regions. Finally, we assessed that these regions could discriminate between BRCA positives from healthy samples with an AUC of 0.95, a sensitivity of 88%, and a specificity of 94.74%. Conclusions Our study emphasizes the importance of tumor cell-derived DNA methylation in BC, reporting a different methylation profile between patients carrying mutations in BRCA1, BRCA2, and wild-type controls. Our minimally invasive approach could allow early cancer diagnosis, assessment of minimal residual disease, and monitoring of response to therapy.
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spelling doaj-art-b4709ad2f11d40968a2b2c09a3828f5f2025-08-20T01:50:38ZengBMCJournal of Translational Medicine1479-58762024-10-0122111210.1186/s12967-024-05734-2Differential methylation of circulating free DNA assessed through cfMeDiP as a new tool for breast cancer diagnosis and detection of BRCA1/2 mutationPiera Grisolia0Rossella Tufano1Clara Iannarone2Antonio De Falco3Francesca Carlino4Cinzia Graziano5Raffaele Addeo6Marianna Scrima7Francesco Caraglia8Anna Ceccarelli9Pier Vitale Nuzzo10Alessia Maria Cossu11Stefano Forte12Raffaella Giuffrida13Michele Orditura14Michele Caraglia15Michele Ceccarelli16Sylvester Comprehensive Cancer Center and Department of Public Health Sciences, Miller School of Medicine, University of MiamiLaboratory of Computational Biology, IRGSLaboratory of Molecular and Precision Oncology, Biogem, IRGSLaboratory of Computational Biology, IRGSDepartment of Precision Medicine, University of Campania “Luigi Vanvitelli”Laboratory of Molecular and Precision Oncology, Biogem, IRGSOncology Unit, S. Giovanni di Dio HospitalLaboratory of Molecular and Precision Oncology, Biogem, IRGSDepartment of Precision Medicine, University of Campania “Luigi Vanvitelli”Medical Oncology, Catholic University of the Sacred HeartDepartment of Pathology and Laboratory Medicine, Weill Cornell MedicineLaboratory of Molecular and Precision Oncology, Biogem, IRGSIOM RicercaIOM RicercaDepartment of Precision Medicine, University of Campania “Luigi Vanvitelli”Laboratory of Molecular and Precision Oncology, Biogem, IRGSSylvester Comprehensive Cancer Center and Department of Public Health Sciences, Miller School of Medicine, University of MiamiAbstract Background Recent studies have highlighted the importance of the cell-free DNA (cfDNA) methylation profile in detecting breast cancer (BC) and its different subtypes. We investigated whether plasma cfDNA methylation, using cell-free Methylated DNA Immunoprecipitation and High-Throughput Sequencing (cfMeDIP-seq), may be informative in characterizing breast cancer in patients with BRCA1/2 germline mutations for early cancer detection and response to therapy. Methods We enrolled 23 BC patients with germline mutation of BRCA1 and BRCA2 genes, 19 healthy controls without BRCA1/2 mutation, and two healthy individuals who carried BRCA1/2 mutations. Blood samples were collected for all study subjects at the diagnosis, and plasma was isolated by centrifugation. Cell-free DNA was extracted from 1 mL of plasma, and cfMeDIP-seq was performed for each sample. Shallow whole genome sequencing was performed on the immuno-precipitated samples. Then, the differentially methylated 300-bp regions (DMRs) between 25 BRCA germline mutation carriers and 19 non-carriers were identified. DMRs were compared with tumor-specific regions from public datasets to perform an unbiased analysis. Finally, two statistical classifiers were trained based on the GLMnet and random forest model to evaluate if the identified DMRs could discriminate BRCA-positive from healthy samples. Results We identified 7,095 hypermethylated and 212 hypomethylated regions in 25 BRCA germline mutation carriers compared to 19 controls. These regions discriminate tumors from healthy samples with high accuracy and sensitivity. We show that the circulating tumor DNA of BRCA1/2 mutant breast cancers is characterized by the hypomethylation of genes involved in DNA repair and cell cycle. We uncovered the TFs associated with these DRMs and identified that proteins of the Erythroblast Transformation Specific (ETS) family are particularly active in the hypermethylated regions. Finally, we assessed that these regions could discriminate between BRCA positives from healthy samples with an AUC of 0.95, a sensitivity of 88%, and a specificity of 94.74%. Conclusions Our study emphasizes the importance of tumor cell-derived DNA methylation in BC, reporting a different methylation profile between patients carrying mutations in BRCA1, BRCA2, and wild-type controls. Our minimally invasive approach could allow early cancer diagnosis, assessment of minimal residual disease, and monitoring of response to therapy.https://doi.org/10.1186/s12967-024-05734-2Breast cancerCell-free DNADMRscfMeDIP-seqBRCA1BRCA2
spellingShingle Piera Grisolia
Rossella Tufano
Clara Iannarone
Antonio De Falco
Francesca Carlino
Cinzia Graziano
Raffaele Addeo
Marianna Scrima
Francesco Caraglia
Anna Ceccarelli
Pier Vitale Nuzzo
Alessia Maria Cossu
Stefano Forte
Raffaella Giuffrida
Michele Orditura
Michele Caraglia
Michele Ceccarelli
Differential methylation of circulating free DNA assessed through cfMeDiP as a new tool for breast cancer diagnosis and detection of BRCA1/2 mutation
Journal of Translational Medicine
Breast cancer
Cell-free DNA
DMRs
cfMeDIP-seq
BRCA1
BRCA2
title Differential methylation of circulating free DNA assessed through cfMeDiP as a new tool for breast cancer diagnosis and detection of BRCA1/2 mutation
title_full Differential methylation of circulating free DNA assessed through cfMeDiP as a new tool for breast cancer diagnosis and detection of BRCA1/2 mutation
title_fullStr Differential methylation of circulating free DNA assessed through cfMeDiP as a new tool for breast cancer diagnosis and detection of BRCA1/2 mutation
title_full_unstemmed Differential methylation of circulating free DNA assessed through cfMeDiP as a new tool for breast cancer diagnosis and detection of BRCA1/2 mutation
title_short Differential methylation of circulating free DNA assessed through cfMeDiP as a new tool for breast cancer diagnosis and detection of BRCA1/2 mutation
title_sort differential methylation of circulating free dna assessed through cfmedip as a new tool for breast cancer diagnosis and detection of brca1 2 mutation
topic Breast cancer
Cell-free DNA
DMRs
cfMeDIP-seq
BRCA1
BRCA2
url https://doi.org/10.1186/s12967-024-05734-2
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