High prevalence of constitutional BRCA1 epimutation in patients with early-onset triple-negative breast cancer

Abstract Between 5 and 8% of the general population carry a constitutional methylation of the BRCA1 promoter (“epimutation”). Several studies have suggested that these BRCA1 epimutations confer an increased risk of breast cancer, in particular triple-negative breast cancer (TNBC), with an earlier on...

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Main Authors: Mathias Schwartz, Sabrina Ibadioune, Hélène Delhomelle, Solenn Barraud, Sandrine M. Caputo, Olfa Trabelsi-Grati, Marie-Charlotte Villy, Anthony Laugé, Roseline Tang, Etienne Rouleau, Emmanuelle Mouret-Fourme, Dominique Stoppa-Lyonnet, Éric Pasmant, Lisa Golmard, Chrystelle Colas, Ivan Bièche
Format: Article
Language:English
Published: BMC 2025-06-01
Series:Clinical Epigenetics
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Online Access:https://doi.org/10.1186/s13148-025-01885-1
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author Mathias Schwartz
Sabrina Ibadioune
Hélène Delhomelle
Solenn Barraud
Sandrine M. Caputo
Olfa Trabelsi-Grati
Marie-Charlotte Villy
Anthony Laugé
Roseline Tang
Etienne Rouleau
Emmanuelle Mouret-Fourme
Dominique Stoppa-Lyonnet
Éric Pasmant
Lisa Golmard
Chrystelle Colas
Ivan Bièche
author_facet Mathias Schwartz
Sabrina Ibadioune
Hélène Delhomelle
Solenn Barraud
Sandrine M. Caputo
Olfa Trabelsi-Grati
Marie-Charlotte Villy
Anthony Laugé
Roseline Tang
Etienne Rouleau
Emmanuelle Mouret-Fourme
Dominique Stoppa-Lyonnet
Éric Pasmant
Lisa Golmard
Chrystelle Colas
Ivan Bièche
author_sort Mathias Schwartz
collection DOAJ
description Abstract Between 5 and 8% of the general population carry a constitutional methylation of the BRCA1 promoter (“epimutation”). Several studies have suggested that these BRCA1 epimutations confer an increased risk of breast cancer, in particular triple-negative breast cancer (TNBC), with an earlier onset than in the general population. However, those studies relied on very few patients with early-onset TNBC. Using specific Methylation-Sensitive High-Resolution Melting, we assessed BRCA1 epimutation prevalence in a large cohort of 112 early-onset (≤ 30 years-old) TNBC patients with no genetic cancer-predisposing pathogenic variants (PVs). We compared this cohort to 87 early-onset TNBC patients carrying cancer-predisposing PVs and to 93 late-onset (≥ 70 years-old) TNBC with no cancer-predisposing PVs. We observed a high prevalence of BRCA1 epimutation in blood cells from early-onset TNBC patients with no cancer-predisposing PVs (38/112, 33.9% [95% confidence interval: 25.4–43.6%]) as compared to early-onset patients with cancer-predisposing PVs (1/87, 1.1% [0.1–7.1%], p value < 0.001) and late-onset patients (11/93, 11.8% [6.3–20.6%], p value < 0.001). These differences remained significant when restricting to epimutations with low variant epiallele frequencies (VEF under 1%). Our results highlight the role of constitutional BRCA1 epimutations in early-onset TNBC risk and call for their integration into multifactorial models used to compute personalized breast cancer risk.
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spelling doaj-art-e303b27e55094aec90ad74aefa2b2f6a2025-08-20T02:30:42ZengBMCClinical Epigenetics1868-70832025-06-011711610.1186/s13148-025-01885-1High prevalence of constitutional BRCA1 epimutation in patients with early-onset triple-negative breast cancerMathias Schwartz0Sabrina Ibadioune1Hélène Delhomelle2Solenn Barraud3Sandrine M. Caputo4Olfa Trabelsi-Grati5Marie-Charlotte Villy6Anthony Laugé7Roseline Tang8Etienne Rouleau9Emmanuelle Mouret-Fourme10Dominique Stoppa-Lyonnet11Éric Pasmant12Lisa Golmard13Chrystelle Colas14Ivan Bièche15Department of Genetics, Curie InstituteDepartment of Genetics, Curie InstituteDepartment of Genetics, Curie InstituteDepartment of Genetics, Curie InstituteDepartment of Genetics, Curie InstituteDepartment of Genetics, Curie InstituteDepartment of Genetics, Curie InstituteDepartment of Genetics, Curie InstituteCancer Genetics Laboratory, Medical Biology and Pathology Department, Gustave-Roussy Cancer CampusCancer Genetics Laboratory, Medical Biology and Pathology Department, Gustave-Roussy Cancer CampusDepartment of Genetics, Curie InstituteDepartment of Genetics, Curie InstituteDepartment of Genetics, Curie InstituteDepartment of Genetics, Curie InstituteDepartment of Genetics, Curie InstituteDepartment of Genetics, Curie InstituteAbstract Between 5 and 8% of the general population carry a constitutional methylation of the BRCA1 promoter (“epimutation”). Several studies have suggested that these BRCA1 epimutations confer an increased risk of breast cancer, in particular triple-negative breast cancer (TNBC), with an earlier onset than in the general population. However, those studies relied on very few patients with early-onset TNBC. Using specific Methylation-Sensitive High-Resolution Melting, we assessed BRCA1 epimutation prevalence in a large cohort of 112 early-onset (≤ 30 years-old) TNBC patients with no genetic cancer-predisposing pathogenic variants (PVs). We compared this cohort to 87 early-onset TNBC patients carrying cancer-predisposing PVs and to 93 late-onset (≥ 70 years-old) TNBC with no cancer-predisposing PVs. We observed a high prevalence of BRCA1 epimutation in blood cells from early-onset TNBC patients with no cancer-predisposing PVs (38/112, 33.9% [95% confidence interval: 25.4–43.6%]) as compared to early-onset patients with cancer-predisposing PVs (1/87, 1.1% [0.1–7.1%], p value < 0.001) and late-onset patients (11/93, 11.8% [6.3–20.6%], p value < 0.001). These differences remained significant when restricting to epimutations with low variant epiallele frequencies (VEF under 1%). Our results highlight the role of constitutional BRCA1 epimutations in early-onset TNBC risk and call for their integration into multifactorial models used to compute personalized breast cancer risk.https://doi.org/10.1186/s13148-025-01885-1HBOCMethylationPromoterCancer predispositionEpimutationConstitutional epimutation
spellingShingle Mathias Schwartz
Sabrina Ibadioune
Hélène Delhomelle
Solenn Barraud
Sandrine M. Caputo
Olfa Trabelsi-Grati
Marie-Charlotte Villy
Anthony Laugé
Roseline Tang
Etienne Rouleau
Emmanuelle Mouret-Fourme
Dominique Stoppa-Lyonnet
Éric Pasmant
Lisa Golmard
Chrystelle Colas
Ivan Bièche
High prevalence of constitutional BRCA1 epimutation in patients with early-onset triple-negative breast cancer
Clinical Epigenetics
HBOC
Methylation
Promoter
Cancer predisposition
Epimutation
Constitutional epimutation
title High prevalence of constitutional BRCA1 epimutation in patients with early-onset triple-negative breast cancer
title_full High prevalence of constitutional BRCA1 epimutation in patients with early-onset triple-negative breast cancer
title_fullStr High prevalence of constitutional BRCA1 epimutation in patients with early-onset triple-negative breast cancer
title_full_unstemmed High prevalence of constitutional BRCA1 epimutation in patients with early-onset triple-negative breast cancer
title_short High prevalence of constitutional BRCA1 epimutation in patients with early-onset triple-negative breast cancer
title_sort high prevalence of constitutional brca1 epimutation in patients with early onset triple negative breast cancer
topic HBOC
Methylation
Promoter
Cancer predisposition
Epimutation
Constitutional epimutation
url https://doi.org/10.1186/s13148-025-01885-1
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