ArticleNature communications2023
Genetic and epigenetic features of bilateral Wilms tumor predisposition in patients from the Children's Oncology Group AREN18B5-Q.
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Article in Nature communications, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 17 papers.
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17citing papers in PubMed
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Who cites it
17 citing papers in PubMed, 24 citations in OpenAlex.
- DNA Methylation and Transcriptomic Profiles of Wilms Tumour Reveal New Deregulated Genes and Epigenetic Processes Relevant for Tumour Stratification and Management.International journal of cancer · 2026Article
- MRPL23 Overexpression Predicts Poor Survival and Is Associated with Mitochondrial Respiratory Signatures in Glioblastoma.Cancers · 2026Article
- Developmental timing distinguishes pediatric and adult cancers through retention and rewiring mechanisms.Nature communications · 2026Review
- Genetic and Epigenetic Drivers of Wilms Tumor Predisposition in Russian Pediatric Patients: A Multicenter Study.International journal of molecular sciences · 2026Article
- Developmental reprogramming underlies chemotherapy resistance in favorable-histology Wilms tumor.Cell reports · 2026Article
- Engineered internal architecture of core-shell lipid nanoparticles promotes efficient mRNA endosomal release.Nature communications · 2026Article
- ALKBH1 Gene rs6494 T>A Polymorphism Decreases Wilms Tumour Risk in Chinese Children.Journal of cellular and molecular medicine · 2025Article
- From preneoplastic lesion to heterogenous tumor: recent insights into hepatoblastoma biology and therapeutic opportunities.Molecular cancer · 2025Review
- Epigenetic Regulation in Wilms Tumor.Biomedicines · 2025Review
- Distinct pathways for genetic and epigenetic predisposition in familial and bilateral Wilms tumor.Genome medicine · 2025Article
- Wilms' Tumor: A Review of Clinical Characteristics, Treatment Advances, and Research Opportunities.Medicina (Kaunas, Lithuania) · 2025Review
- Re-envisioning genetic predisposition to childhood and adolescent cancers.Nature reviews. Cancer · 2025Review
- NK cell-related genes-driven novel molecular subtyping and prognostic signatures for Wilms tumor: uncovering the therapeutic potential of TGX-221 and biomarker role of HS2ST1.Frontiers in oncology · 2025Article
- Pursuit of the optimal therapeutic approach and intensity for children with bilateral Wilms tumour.British journal of cancer · 2024Article
- Genomic Newborn Screening for Pediatric Cancer Predisposition Syndromes: A Holistic Approach.Cancers · 2024Review
- Characterization of Alternative Splicing in High-Risk Wilms' Tumors.International journal of molecular sciences · 2024Article
- Bilateral Wilms Tumor - Case Report of a Patient with Family History.The application of clinical genetics · 2024Article
Corrections and comments
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Authors and funding
34 authors at 13 institutions in 2 countries.
Andrew J Murphy *Department of Surgery, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA. andrew.murphy@stjude.org.ORCID 0000-0001-6747-0355
Changde Cheng *Department of Computational Biology, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.ORCID 0000-0002-2458-2522
Justin WilliamsDepartment of Computational Biology, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.
Timothy I ShawDepartment of Computational Biology, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.ORCID 0000-0002-9316-1924
Emilia M PintoDepartment of Pathology, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.ORCID 0000-0001-5567-000X
Karissa Dieseldorff-JonesDepartment of Computational Biology, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.ORCID 0000-0002-9124-6246
Jack BrzezinskiDepartment of Oncology, The Hospital for Sick Children, Toronto, ON, Canada.
Lindsay A RenfroChildren's Oncology Group and Department of Population and Public Health Sciences, Keck School of Medicine of University of Southern California, Los Angeles, CA, USA.
Brett TornwallChildren's Oncology Group Statistics and Data Center, Monrovia, CA, USA.
Vicki HuffDepartment of Genetics, The University of Texas MD Anderson Cancer Center, Houston, TX, USA.
Andrew L HongDepartment of Pediatrics, Emory University School of Medicine, Atlanta, GA, USA.ORCID 0000-0003-0374-1667
Elizabeth A MullenDepartment of Pediatric Oncology, Dana-Farber/Boston Children's Cancer and Blood Disorders Center and Harvard Medical School, Boston, MA, 02215, USA.ORCID 0000-0002-6704-0002
Brian CromptonDepartment of Pediatric Oncology, Dana-Farber/Boston Children's Cancer and Blood Disorders Center and Harvard Medical School, Boston, MA, 02215, USA.ORCID 0000-0001-9404-6621
Jeffrey S DomeCenter for Cancer and Blood Disorders, Children's National Hospital, Department of Pediatrics, George Washington University School of Medicine and Health Sciences, Washington, DC, USA.ORCID 0000-0002-9037-369X
Conrad V FernandezIWK Health Center and Dalhousie University, Halifax, NS, Canada.ORCID 0000-0002-0746-8306
James I GellerDivision of Oncology, Cincinnati Children's Hospital Medical Center, University of Cincinnati, Cincinnati, OH, USA.
Peter F EhrlichSection of Pediatric Surgery, C.S. Mott Children's Hospital, University of Michigan, Ann Arbor, MI, USA.
Heather MulderDepartment of Computational Biology, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.ORCID 0000-0003-2024-9498
Ninad OakDepartment of Oncology, St. Jude Children's Research Hospital, Memphis, TN, USA.ORCID 0000-0002-9806-0217
Jamie MaciezsekDepartment of Pathology, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.
Carolyn M JablonowskiDepartment of Surgery, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.
Andrew M FlemingDepartment of Surgery, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.ORCID 0000-0001-5421-9014
Prahalathan PichavaramDepartment of Surgery, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.
Christopher L MortonDepartment of Surgery, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.
John EastonDepartment of Computational Biology, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.ORCID 0000-0003-4503-6608
Kim E NicholsDepartment of Oncology, St. Jude Children's Research Hospital, Memphis, TN, USA.ORCID 0000-0002-5581-6555
Michael R ClayDepartment of Pathology, University of Colorado Anschutz, Aurora, CO, USA.
Teresa SantiagoDepartment of Pathology, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.
Jinghui ZhangDepartment of Computational Biology, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.ORCID 0000-0003-3350-9682
Jun YangDepartment of Surgery, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.ORCID 0000-0002-0770-9659
Gerard P ZambettiDepartment of Pathology, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.ORCID 0000-0002-0929-5007
Zhaoming WangDepartment of Computational Biology, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.ORCID 0000-0001-7556-3869
Andrew M DavidoffDepartment of Surgery, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA.ORCID 0000-0001-7900-2794
Xiang ChenDepartment of Computational Biology, St. Jude Children's Research Hospital, Memphis, TN, 38105, USA. Xiang.chen@stjude.org.ORCID 0000-0002-2499-8261
St. Jude Children's Research Hospital · USBroad Institute · USChildren's Oncology Group · USCincinnati Children's Hospital Medical Center · USDalhousie University · CAEmory University · USGeorge Washington University · USHarvard University · USHospital for Sick Children · CAThe University of Texas MD Anderson Cancer Center · USUniversity of Colorado Anschutz Medical Campus · USUniversity of Michigan · USUniversity of Southern California · US
Funding
NCTN BIQSFP ANBL1531 (NRT)U10CA180886 · NCI · PUBLIC HEALTH INSTITUTE · PI Douglas S. Hawkins · 2014 to 2026
$390.6MCOG FOREIGN ACCRUALU10CA098543 · NCI · NATIONAL CHILDHOOD CANCER FOUNDATION · PI ADAMSON, PETER C. · 2003 to 2013
$335.5MViral Vector Technology (VVTSR)P30CA021765 · NCI · ST. JUDE CHILDREN'S RESEARCH HOSPITAL · PI Gregory Armstrong · 1985 to 2026
$166.9MCOG SDMC - Statistics CoreU10CA180899 · NCI · UNIVERSITY OF SOUTHERN CALIFORNIA · PI TODD A ALONZO · 2014 to 2026
$132.8MChildren's Oncology Group Statistics &Data Center GrantU10CA098413 · NCI · UNIVERSITY OF NEBRASKA MEDICAL CENTER · PI ANDERSON, JAMES R · 2003 to 2013
$67.5MSupport for Human Specimen Banking in NCI-Supported Cancer Clinical TrialsU24CA114766 · NCI · RESEARCH INST NATIONWIDE CHILDREN'S HOSP · PI RAMIREZ MILAN, NILSA DEL CARMEN · 2005 to 2014
$14.5MEXPLORE AND TARGET THE EPIGENETIC VULNERABILITY OF PAX3-FOXO1-DRIVEN RHABDOMYOSARCOMAR01CA266600 · NCI · ST. JUDE CHILDREN'S RESEARCH HOSPITAL · PI Xiang Chen, Jun Yang · 2022 to 2026
$3.4MDevelopment of small molecules to target KDM4BR01CA229739 · NCI · ST. JUDE CHILDREN'S RESEARCH HOSPITAL · PI CHEN, TAOSHENG, DAVIDOFF, ANDREW M · 2018 to 2021
$1.6MWT1 as an oncogene and therapeutic target in anaplastic Wilms tumorK08CA255569 · NCI · ST. JUDE CHILDREN'S RESEARCH HOSPITAL · PI MURPHY, ANDREW J · 2021 to 2025
$1.2MDevelopment, review, and conduct of Children's Oncology Group (COG) Clinical TrialsR50CA275931 · NCI · CHILDREN'S RESEARCH INSTITUTE · PI JEFFREY S DOME · 2023 to 2026
$569kNCI NIH HHS K08 CA255569NCI NIH HHS L40 CA242492NCI NIH HHS P30 CA021765NCI NIH HHS R01 CA229739NCI NIH HHS R01 CA266600NCI NIH HHS R50 CA275931NCI NIH HHS U10 CA098413NCI NIH HHS U10 CA098543NCI NIH HHS U10 CA180886NCI NIH HHS U10 CA180899NCI NIH HHS U24 CA114766
Abstract
Developing synchronous bilateral Wilms tumor suggests an underlying (epi)genetic predisposition. Here, we evaluate this predisposition in 68 patients using whole exome or genome sequencing (n = 85 tumors from 61 patients with matched germline blood DNA), RNA-seq (n = 99 tumors), and DNA methylation analysis (n = 61 peripheral blood, n = 29 non-diseased kidney, n = 99 tumors). We determine the predominant events for bilateral Wilms tumor predisposition: 1)pre-zygotic germline genetic variants readily detectable in blood DNA [WT1 (14.8%), NYNRIN (6.6%), TRIM28 (5%), and BRCA-related genes (5%)] or 2)post-zygotic epigenetic hypermethylation at 11p15.5 H19/ICR1 that may require analysis of multiple tissue types for diagnosis. Of 99 total tumor specimens, 16 (16.1%) have 11p15.5 normal retention of imprinting, 25 (25.2%) have 11p15.5 copy neutral loss of heterozygosity, and 58 (58.6%) have 11p15.5 H19/ICR1 epigenetic hypermethylation (loss of imprinting). Here, we ascertain the epigenetic and genetic modes of bilateral Wilms tumor predisposition.
Indexed as
Kidney NeoplasmsWilms TumorChildDNADNA MethylationEpigenesis, GeneticGenomic ImprintingGenotypeHumansDNA
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