Evidence map›Paper›PMID 41381884›Full record

ArticleNature genetics2026

Investigation of a global mouse methylome atlas reveals subtype-specific copy number alterations in pediatric cancer models.

Melanie Schoof, Tuyu Zheng, Martin Sill, Roland Imle, Alessia Cais, Lea Altendorf, Alicia Fürst, Nina Hofmann, Kati Ernst, Dominik Vonficht and 44 more

Abstract read
In one paragraph

Article in Nature genetics, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

0numbers the graph read from it
0cells of the map it votes in
2citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

2 citing papers in PubMed.

  1. Article
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4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

54 authors.

Melanie Schoof *Research Institute Children's Cancer Center Hamburg, Hamburg, Germany.ORCID 0000-0002-1373-8130
Tuyu Zheng *Division of Pediatric Neurooncology, Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.ORCID 0000-0003-3385-7353
Martin SillDivision of Pediatric Neurooncology, Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.ORCID 0000-0001-7616-7665
Roland ImleDivision of Pediatric Neurooncology, Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.ORCID 0000-0001-9564-6952
Alessia CaisDivision of Pediatric Neurooncology, Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.
Lea AltendorfResearch Institute Children's Cancer Center Hamburg, Hamburg, Germany.
Alicia FürstResearch Institute Children's Cancer Center Hamburg, Hamburg, Germany.
Nina HofmannDivision of Pediatric Neurooncology, Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.
Kati ErnstDivision of Pediatric Neurooncology, Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.ORCID 0000-0002-8555-5907
Dominik VonfichtHeidelberg Institute for Stem Cell Technology and Experimental Medicine (HI-STEM gGmbH), Heidelberg, Germany.
Kenneth Chun-Ho ChanDivision of Pediatric Neurooncology, Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.
Tim Holland-LetzDepartment of Biostatistics, German Cancer Research Center (DKFZ), Heidelberg, Germany.
Andreas PostlmayrTranslational Brain Tumor Research Group, Children's Research Center, University Children's Hospital Zurich, Zurich, Switzerland.
Ryo ShiraishiDepartment of Biochemistry and Cellular Biology, National Institute of Neuroscience, NCNP, Tokyo, Japan.
Wanchen WangDepartment of Biochemistry and Cellular Biology, National Institute of Neuroscience, NCNP, Tokyo, Japan.
Alaide MorcavalloDivision of Clinical Studies, The Institute of Cancer Research, London, UK.
Michael SpohnResearch Institute Children's Cancer Center Hamburg, Hamburg, Germany.ORCID 0000-0001-9160-4157
Carolin GöbelResearch Institute Children's Cancer Center Hamburg, Hamburg, Germany.ORCID 0000-0002-7345-2089
Judith NiesenResearch Institute Children's Cancer Center Hamburg, Hamburg, Germany.
Levke-Sophie PeterResearch Institute Children's Cancer Center Hamburg, Hamburg, Germany.ORCID 0009-0005-7283-9061
Franck BourdeautSIREDO Pediatric Oncology Center, Institut Curie, Paris-Science Lettres University, Paris, France.ORCID 0000-0001-9489-6781
Zhi-Yan HanSIREDO Pediatric Oncology Center, Institut Curie, Paris-Science Lettres University, Paris, France.
Yanxin PeiCenter for Cancer and Immunology, Brain Tumor Institute, Children's National Health System, Washington, DC, USA.
Najiba MuradCenter for Cancer and Immunology, Brain Tumor Institute, Children's National Health System, Washington, DC, USA.
Fredrik J SwartlingDepartment of Immunology, Genetics and Pathology, Uppsala University, Uppsala, Sweden.ORCID 0000-0002-8460-4367
Jessica TaylorCancer Research UK Cambridge Institute, Li Ka Shing Centre, University of Cambridge, Cambridge, UK.ORCID 0000-0001-8565-8589
Monika YadavDepartment of Pediatrics, Children's Mercy Research Institute (CMRI), Kansas City, MO, USA.
Garrett R GibsonDepartment of Pediatrics, Children's Mercy Research Institute (CMRI), Kansas City, MO, USA.
Richard J GilbertsonCancer Research UK Cambridge Institute, Li Ka Shing Centre, University of Cambridge, Cambridge, UK.ORCID 0000-0001-7539-9472
Matthias DottermuschInstitute of Neuropathology, University Medical Center Hamburg-Eppendorf, Hamburg, Germany.
Rajanya RoyDepartment of Pediatric Hematology and Oncology, University Children's Hospital Münster, Münster, Germany.
Kornelius KerlDepartment of Pediatric Hematology and Oncology, University Children's Hospital Münster, Münster, Germany.ORCID 0000-0002-5676-8102
Rainer GlassNeurosurgical Research, Department of Neurosurgery, LMU University Hospital, LMU Munich, München, Germany.
Jiying ChengNeurosurgical Research, Department of Neurosurgery, LMU University Hospital, LMU Munich, München, Germany.
Martin A HorstmannResearch Institute Children's Cancer Center Hamburg, Hamburg, Germany.
Gerrit Wolters-EisfeldResearch Institute Children's Cancer Center Hamburg, Hamburg, Germany.ORCID 0000-0002-2984-6515
Haotian ZhaoDepartment of Biomedical Sciences, New York Institute of Technology College of Osteopathic Medicine, Old Westbury, NY, USA.ORCID 0000-0003-2315-8452
Dominik SturmNational Center for Tumor Diseases (NCT) Heidelberg, a partnership between DKFZ and Heidelberg University Hospital, Heidelberg, Germany.
Viveka Nand YadavDepartment of Pediatrics, Children's Mercy Research Institute (CMRI), Kansas City, MO, USA.
Louis CheslerPaediatric Oncology Experimental Medicine Centre, The Institute of Cancer Research, London, UK.ORCID 0000-0001-7842-2068
Simon HaasBerlin Institute of Health (BIH) at Charité Universitätsmedizin Berlin, Berlin, Germany.ORCID 0000-0001-9227-2051
William A WeissUCSF Helen Diller Family Comprehensive Cancer Center, San Francisco, CA, USA.
Paul A NorthcottCenter of Excellence in Neuro-Oncology Sciences, St. Jude Children's Research Hospital, Memphis, TN, USA.ORCID 0000-0002-1220-5252
Lena M KutscherDivision of Pediatric Neurooncology, Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.ORCID 0000-0002-1130-4582
Ana Guerreiro StucklinTranslational Brain Tumor Research Group, Children's Research Center, University Children's Hospital Zurich, Zurich, Switzerland.ORCID 0000-0003-3136-9241
Olivier AyraultInstitut Curie, PSL Research University, CNRS UMR, INSERM, Orsay, France.ORCID 0000-0002-7942-6674
Julia E NeumannInstitute of Neuropathology, University Medical Center Hamburg-Eppendorf, Hamburg, Germany.ORCID 0000-0002-1162-8771
Daisuke KawauchiDepartment of Biochemistry and Cellular Biology, National Institute of Neuroscience, NCNP, Tokyo, Japan.
David T W JonesNational Center for Tumor Diseases (NCT) Heidelberg, a partnership between DKFZ and Heidelberg University Hospital, Heidelberg, Germany.ORCID 0000-0002-2036-5141
Kristian PajtlerDivision of Pediatric Neurooncology, Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.
Ana BanitoDivision of Pediatric Neurooncology, Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.ORCID 0000-0003-2188-0003
Stefan M PfisterDivision of Pediatric Neurooncology, Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany.ORCID 0000-0002-5447-5322
Ulrich SchüllerResearch Institute Children's Cancer Center Hamburg, Hamburg, Germany.ORCID 0000-0002-8731-1121
Marc ZuckermannDivision of Pediatric Neurooncology, Hopp Children's Cancer Center Heidelberg (KiTZ), Heidelberg, Germany. m.zuckermann@kitz-heidelberg.de.ORCID 0000-0003-0148-3289

Funding

(PQ1) Molecular circuit of multi-ciliogenesis regulates choroid plexus differentiation and tumor developmentR01CA220551 · NCI · SANFORD RESEARCH/USD · PI ZHAO, HAOTIAN · 2017 to 2021
$1.7M
MicroRNA 34/449 family plays a critical role in choroid plexus differentiation and tumorigenesisR15CA287297 · NCI · NEW YORK INST OF TECHNOLOGY · PI ZHAO, HAOTIAN · 2025 to 2025
$459k
Deutsche Forschungsgemeinschaft (German Research Foundation) SFB 1389EC | EU Framework Programme for Research and Innovation H2020 | H2020 Priority Excellent Science | H2020 European Research Council (H2020 Excellent Science - European Research Council) 101117088EC | EU Framework Programme for Research and Innovation H2020 | H2020 Priority Excellent Science | H2020 European Research Council (H2020 Excellent Science - European Research Council) 819894NCI NIH HHS R01 CA220551NCI NIH HHS R15 CA287297
6 · The paper itself

Abstract

Copy number alterations (CNAs) are hallmarks of cancer, yet investigation of their oncogenic role has been hindered by technical limitations and missing model systems. Here we generated a genome-wide DNA methylation and CNA atlas of 106 genetic mouse models across 31 pediatric tumor types, including 18 new models for pediatric glioma. We demonstrated their epigenetic resemblance to human disease counterparts and identified entity-specific patterns of immune infiltration. We discovered that mouse tumors harbor highly recurrent CNA signatures that occur distinctly based on the tumor subgroup and driving oncogene and showed that these CNAs share syntenic regions with the matching human tumor types, thereby revealing a conserved but previously underappreciated role in subgroup-specific tumorigenesis that can be analyzed using the presented models. Our study provides insights into globally available mouse models for pediatric solid cancers and enables access to functional CNA interrogation, with the potential to unlock new translational targets in pediatric cancers.

Indexed as

DNA Copy Number VariationsDNA MethylationEpigenomeNeoplasmsAnimalsChildDisease Models, AnimalEpigenesis, GeneticGene Expression Regulation, NeoplasticHumansMice

Identifiers

PMID41381884
PMCPMC12807868

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