Endometrial tumorigenesis involves epigenetic plasticity demarcating non-coding somatic mutations and 3D-genome alterations

Abstract Background The incidence and mortality of endometrial cancer (EC) is on the rise. Eighty-five percent of ECs depend on estrogen receptor alpha (ERα) for proliferation, but little is known about its transcriptional regulation in these tumors. Results We generate epigenomics, transcriptomics,...

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Main Authors: Sebastian Gregoricchio, Aleksandar Kojic, Marlous Hoogstraat, Karianne Schuurman, Suzan Stelloo, Tesa M. Severson, Tracy A. O’Mara, Marjolein Droog, Abhishek A. Singh, Dylan M. Glubb, Lodewyk F. A. Wessels, Michiel Vermeulen, Flora E. van Leeuwen, Wilbert Zwart
Format: Article
Language:English
Published: BMC 2025-05-01
Series:Genome Biology
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Online Access:https://doi.org/10.1186/s13059-025-03596-5
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author Sebastian Gregoricchio
Aleksandar Kojic
Marlous Hoogstraat
Karianne Schuurman
Suzan Stelloo
Tesa M. Severson
Tracy A. O’Mara
Marjolein Droog
Abhishek A. Singh
Dylan M. Glubb
Lodewyk F. A. Wessels
Michiel Vermeulen
Flora E. van Leeuwen
Wilbert Zwart
author_facet Sebastian Gregoricchio
Aleksandar Kojic
Marlous Hoogstraat
Karianne Schuurman
Suzan Stelloo
Tesa M. Severson
Tracy A. O’Mara
Marjolein Droog
Abhishek A. Singh
Dylan M. Glubb
Lodewyk F. A. Wessels
Michiel Vermeulen
Flora E. van Leeuwen
Wilbert Zwart
author_sort Sebastian Gregoricchio
collection DOAJ
description Abstract Background The incidence and mortality of endometrial cancer (EC) is on the rise. Eighty-five percent of ECs depend on estrogen receptor alpha (ERα) for proliferation, but little is known about its transcriptional regulation in these tumors. Results We generate epigenomics, transcriptomics, and Hi-C datastreams in healthy and tumor endometrial tissues, identifying robust ERα reprogramming and profound alterations in 3D genome organization that lead to a gain of tumor-specific enhancer activity during EC development. Integration with endometrial cancer risk single-nucleotide polymorphisms and whole-genome sequencing data from primary tumors and metastatic samples reveals a striking enrichment of risk variants and non-coding somatic mutations at tumor-enriched ERα sites. Through machine learning-based predictions and interaction proteomics analyses, we identify an enhancer mutation which alters 3D genome conformation, impairing recruitment of the transcriptional repressor EHMT2/G9a/KMT1C, thereby alleviating transcriptional repression of ESR1 in EC. Conclusions In summary, we identify a complex genomic-epigenomic interplay in EC development and progression, altering 3D genome organization to enhance expression of the critical driver ERα.
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issn 1474-760X
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spelling doaj-art-b545d677e4464fa18aa71d22dc97fb392025-08-20T02:32:03ZengBMCGenome Biology1474-760X2025-05-0126113110.1186/s13059-025-03596-5Endometrial tumorigenesis involves epigenetic plasticity demarcating non-coding somatic mutations and 3D-genome alterationsSebastian Gregoricchio0Aleksandar Kojic1Marlous Hoogstraat2Karianne Schuurman3Suzan Stelloo4Tesa M. Severson5Tracy A. O’Mara6Marjolein Droog7Abhishek A. Singh8Dylan M. Glubb9Lodewyk F. A. Wessels10Michiel Vermeulen11Flora E. van Leeuwen12Wilbert Zwart13Division of Oncogenomics, Oncode Institute, The Netherlands Cancer InstituteDivision of Oncogenomics, Oncode Institute, The Netherlands Cancer InstituteDivision of Oncogenomics, Oncode Institute, The Netherlands Cancer InstituteDivision of Oncogenomics, Oncode Institute, The Netherlands Cancer InstituteDepartment of Molecular Biology, Faculty of Science, Radboud Institute for Molecular Life Sciences, Oncode Institute, Radboud University NijmegenDivision of Oncogenomics, Oncode Institute, The Netherlands Cancer InstituteCancer Research Program, QIMR Berghofer Medical Research InstituteDivision of Oncogenomics, Oncode Institute, The Netherlands Cancer InstituteDivision of Oncogenomics, Oncode Institute, The Netherlands Cancer InstituteCancer Research Program, QIMR Berghofer Medical Research InstituteDivision of Molecular Carcinogenesis, Oncode Institute, The Netherlands Cancer InstituteDepartment of Molecular Biology, Faculty of Science, Radboud Institute for Molecular Life Sciences, Oncode Institute, Radboud University NijmegenDepartment of Epidemiology, The Netherlands Cancer InstituteDivision of Oncogenomics, Oncode Institute, The Netherlands Cancer InstituteAbstract Background The incidence and mortality of endometrial cancer (EC) is on the rise. Eighty-five percent of ECs depend on estrogen receptor alpha (ERα) for proliferation, but little is known about its transcriptional regulation in these tumors. Results We generate epigenomics, transcriptomics, and Hi-C datastreams in healthy and tumor endometrial tissues, identifying robust ERα reprogramming and profound alterations in 3D genome organization that lead to a gain of tumor-specific enhancer activity during EC development. Integration with endometrial cancer risk single-nucleotide polymorphisms and whole-genome sequencing data from primary tumors and metastatic samples reveals a striking enrichment of risk variants and non-coding somatic mutations at tumor-enriched ERα sites. Through machine learning-based predictions and interaction proteomics analyses, we identify an enhancer mutation which alters 3D genome conformation, impairing recruitment of the transcriptional repressor EHMT2/G9a/KMT1C, thereby alleviating transcriptional repression of ESR1 in EC. Conclusions In summary, we identify a complex genomic-epigenomic interplay in EC development and progression, altering 3D genome organization to enhance expression of the critical driver ERα.https://doi.org/10.1186/s13059-025-03596-5Endometrial cancerEstrogen receptorEpigenetic plasticity in tumor developmentGene regulation3D genome organizationNon-coding somatic mutations
spellingShingle Sebastian Gregoricchio
Aleksandar Kojic
Marlous Hoogstraat
Karianne Schuurman
Suzan Stelloo
Tesa M. Severson
Tracy A. O’Mara
Marjolein Droog
Abhishek A. Singh
Dylan M. Glubb
Lodewyk F. A. Wessels
Michiel Vermeulen
Flora E. van Leeuwen
Wilbert Zwart
Endometrial tumorigenesis involves epigenetic plasticity demarcating non-coding somatic mutations and 3D-genome alterations
Genome Biology
Endometrial cancer
Estrogen receptor
Epigenetic plasticity in tumor development
Gene regulation
3D genome organization
Non-coding somatic mutations
title Endometrial tumorigenesis involves epigenetic plasticity demarcating non-coding somatic mutations and 3D-genome alterations
title_full Endometrial tumorigenesis involves epigenetic plasticity demarcating non-coding somatic mutations and 3D-genome alterations
title_fullStr Endometrial tumorigenesis involves epigenetic plasticity demarcating non-coding somatic mutations and 3D-genome alterations
title_full_unstemmed Endometrial tumorigenesis involves epigenetic plasticity demarcating non-coding somatic mutations and 3D-genome alterations
title_short Endometrial tumorigenesis involves epigenetic plasticity demarcating non-coding somatic mutations and 3D-genome alterations
title_sort endometrial tumorigenesis involves epigenetic plasticity demarcating non coding somatic mutations and 3d genome alterations
topic Endometrial cancer
Estrogen receptor
Epigenetic plasticity in tumor development
Gene regulation
3D genome organization
Non-coding somatic mutations
url https://doi.org/10.1186/s13059-025-03596-5
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