Long-read sequencing of diagnosis and post-therapy medulloblastoma reveals complex rearrangement patterns and epigenetic signatures.

Nanopore methylation calling cancer genomics chromothripsis complex rearrangements epigenetic signatures long read sequencing templated insertions

Journal

Cell genomics
ISSN: 2666-979X
Titre abrégé: Cell Genom
Pays: United States
ID NLM: 9918284260106676

Informations de publication

Date de publication:
12 Apr 2023
Historique:
received: 20 02 2022
revised: 14 06 2022
accepted: 22 02 2023
medline: 21 4 2023
pubmed: 21 4 2023
entrez: 21 04 2023
Statut: epublish

Résumé

Cancer genomes harbor a broad spectrum of structural variants (SVs) driving tumorigenesis, a relevant subset of which escape discovery using short-read sequencing. We employed Oxford Nanopore Technologies (ONT) long-read sequencing in a paired diagnostic and post-therapy medulloblastoma to unravel the haplotype-resolved somatic genetic and epigenetic landscape. We assembled complex rearrangements, including a 1.55-Mbp chromothripsis event, and we uncover a complex SV pattern termed templated insertion (TI) thread, characterized by short (mostly <1 kb) insertions showing prevalent self-concatenation into highly amplified structures of up to 50 kbp in size. TI threads occur in 3% of cancers, with a prevalence up to 74% in liposarcoma, and frequent colocalization with chromothripsis. We also perform long-read-based methylome profiling and discover allele-specific methylation (ASM) effects, complex rearrangements exhibiting differential methylation, and differential promoter methylation in cancer-driver genes. Our study shows the advantage of long-read sequencing in the discovery and characterization of complex somatic rearrangements.

Identifiants

pubmed: 37082141
doi: 10.1016/j.xgen.2023.100281
pii: S2666-979X(23)00041-1
pmc: PMC10112291
doi:

Types de publication

Journal Article

Langues

eng

Pagination

100281

Informations de copyright

© 2023 The Authors.

Déclaration de conflit d'intérêts

E.B. is a paid consultant and shareholder of ONT. A.L. has received financial support from ONT for consumables during the course of the project and is currently an employee of ONT. O.S. is a paid consultant of Insitro, Inc.

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Auteurs

Tobias Rausch (T)

European Molecular Biology Laboratory (EMBL), Genome Biology Unit, Heidelberg, Germany.
European Molecular Biology Laboratory (EMBL), GeneCore, Heidelberg, Germany.

Rene Snajder (R)

Division of Computational Genomics and Systems Genetics, German Cancer Research Center (DKFZ), Heidelberg, Germany.
Faculty for Biosciences, Heidelberg University, Heidelberg, Germany.
HIDSS4Health, Helmholtz Information and Data Science School for Health, Heidelberg, Germany.

Adrien Leger (A)

European Molecular Biology Laboratory (EMBL), European Bioinformatics Institute, Wellcome Genome Campus, Hinxton, Cambridge, UK.

Milena Simovic (M)

Group "Genome Instability in Tumors," German Cancer Research Center (DKFZ), Heidelberg, Germany.

Mădălina Giurgiu (M)

Experimental and Clinical Research Center (ECRC) of the Max Delbrück Center (MDC) and Charité-Universitätsmedizin, Berlin, Germany.
Freie Universität Berlin, Berlin, Germany.

Laura Villacorta (L)

European Molecular Biology Laboratory (EMBL), GeneCore, Heidelberg, Germany.

Anton G Henssen (AG)

Department of Pediatric Oncology/Hematology, Charité-Universitätsmedizin, Berlin, Germany.
Experimental and Clinical Research Center (ECRC) of the Max Delbrück Center (MDC) and Charité-Universitätsmedizin, Berlin, Germany.
German Cancer Consortium (DKTK), partner site Berlin, and German Cancer Research Center (DKFZ), Heidelberg, Germany.

Stefan Fröhling (S)

National Center for Tumor Diseases (NCT), Heidelberg, Germany.
German Cancer Research Center (DKFZ), Heidelberg, Germany.
German Cancer Consortium (DKTK), Heidelberg, Germany.

Oliver Stegle (O)

European Molecular Biology Laboratory (EMBL), Genome Biology Unit, Heidelberg, Germany.
Division of Computational Genomics and Systems Genetics, German Cancer Research Center (DKFZ), Heidelberg, Germany.
Wellcome Sanger Institute, Wellcome Trust Genome Campus, Cambridge, UK.

Ewan Birney (E)

European Molecular Biology Laboratory (EMBL), European Bioinformatics Institute, Wellcome Genome Campus, Hinxton, Cambridge, UK.

Marc Jan Bonder (MJ)

Division of Computational Genomics and Systems Genetics, German Cancer Research Center (DKFZ), Heidelberg, Germany.

Aurelie Ernst (A)

Group "Genome Instability in Tumors," German Cancer Research Center (DKFZ), Heidelberg, Germany.

Jan O Korbel (JO)

European Molecular Biology Laboratory (EMBL), Genome Biology Unit, Heidelberg, Germany.
European Molecular Biology Laboratory (EMBL), European Bioinformatics Institute, Wellcome Genome Campus, Hinxton, Cambridge, UK.
Bridging Research Division on Mechanisms of Genomic Variation and Data Science, DKFZ, Heidelberg, Germany.

Classifications MeSH