Frequency of pathogenic germline variants in pediatric medulloblastoma survivors.

germline medulloblastoma pathogenic pediatric survivor

Journal

Frontiers in oncology
ISSN: 2234-943X
Titre abrégé: Front Oncol
Pays: Switzerland
ID NLM: 101568867

Informations de publication

Date de publication:
2024
Historique:
received: 31 05 2024
accepted: 17 07 2024
medline: 26 8 2024
pubmed: 26 8 2024
entrez: 26 8 2024
Statut: epublish

Résumé

Medulloblastoma is the most common malignant brain tumor in children. Most cases are sporadic, but well characterized germline alterations in Germline exome sequencing was conducted on 160 childhood survivors of medulloblastoma. Analyses focused on rare variants in 239 known cancer susceptibility genes (CSGs). P/LP variants were identified using ClinVar and InterVar. Variants of unknown significance in known medulloblastoma predisposing genes ( Twenty cases (12.5%) had a P/LP variant in an autosomal dominant CSG versus 5% in controls (p=1.0 x10 Approximately one in eight pediatric medulloblastoma survivors had an autosomal dominant P/LP CSG variant. We confirm several known associated genes and identify novel genes that may be important in medulloblastoma.

Sections du résumé

Background UNASSIGNED
Medulloblastoma is the most common malignant brain tumor in children. Most cases are sporadic, but well characterized germline alterations in
Methods UNASSIGNED
Germline exome sequencing was conducted on 160 childhood survivors of medulloblastoma. Analyses focused on rare variants in 239 known cancer susceptibility genes (CSGs). P/LP variants were identified using ClinVar and InterVar. Variants of unknown significance in known medulloblastoma predisposing genes (
Results UNASSIGNED
Twenty cases (12.5%) had a P/LP variant in an autosomal dominant CSG versus 5% in controls (p=1.0 x10
Conclusion UNASSIGNED
Approximately one in eight pediatric medulloblastoma survivors had an autosomal dominant P/LP CSG variant. We confirm several known associated genes and identify novel genes that may be important in medulloblastoma.

Identifiants

pubmed: 39184053
doi: 10.3389/fonc.2024.1441958
pmc: PMC11341988
doi:

Types de publication

Journal Article

Langues

eng

Pagination

1441958

Informations de copyright

Copyright © 2024 Rees, Gianferante, Kim, Stavrou, Reaman, Sapkota, Gramatges, Morton, Hudson, Armstrong, Freedman, Huang, Diver, Lori, Luo, Hicks, Liu, Hutchinson, Goldstein and Mirabello.

Déclaration de conflit d'intérêts

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Auteurs

Donald Rees (D)

Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health (NIH), Rockville, MD, United States.
Department of Pediatric Hematology and Oncology, Walter Reed National Military Medical Center, Bethesda, MD, United States.
Department of Pediatrics, Uniformed Services University of the Health Sciences, Bethesda, MD, United States.

D Matthew Gianferante (DM)

Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health (NIH), Rockville, MD, United States.
Department of Pediatrics, Uniformed Services University of the Health Sciences, Bethesda, MD, United States.

Jung Kim (J)

Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health (NIH), Rockville, MD, United States.

Theodora Stavrou (T)

Department of Public Health, Ministry of Health, Athens, Greece.

Gregory Reaman (G)

Division Cancer Treatment and Diagnosis, National Cancer Institute, National Institutes of Health (NIH), Bethesda, MD, United States.

Yadav Sapkota (Y)

Departments of Oncology and Epidemiology and Cancer Control, St. Jude Children's Research Hospital, Memphis, TN, United States.

M Monica Gramatges (MM)

Division of Hematology and Oncology, Department of Pediatrics, Texas Children's Hospital, Baylor College of Medicine, Houston, TX, United States.

Lindsay M Morton (LM)

Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health (NIH), Rockville, MD, United States.

Melissa M Hudson (MM)

Departments of Oncology and Epidemiology and Cancer Control, St. Jude Children's Research Hospital, Memphis, TN, United States.

Gregory T Armstrong (GT)

Departments of Oncology and Epidemiology and Cancer Control, St. Jude Children's Research Hospital, Memphis, TN, United States.

Neal D Freedman (ND)

Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health (NIH), Rockville, MD, United States.

Wen-Yi Huang (WY)

Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health (NIH), Rockville, MD, United States.

W Ryan Diver (WR)

Department of Population Science, American Cancer Society, Atlanta, GA, United States.

Adriana Lori (A)

Department of Population Science, American Cancer Society, Atlanta, GA, United States.

Wen Luo (W)

Cancer Genomics Research Laboratory, Frederick National Laboratory for Cancer Research, Frederick, MD, United States.

Belynda D Hicks (BD)

Cancer Genomics Research Laboratory, Frederick National Laboratory for Cancer Research, Frederick, MD, United States.

Jia Liu (J)

Cancer Genomics Research Laboratory, Frederick National Laboratory for Cancer Research, Frederick, MD, United States.

Amy A Hutchinson (AA)

Cancer Genomics Research Laboratory, Frederick National Laboratory for Cancer Research, Frederick, MD, United States.

Alisa M Goldstein (AM)

Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health (NIH), Rockville, MD, United States.

Lisa Mirabello (L)

Division of Cancer Epidemiology and Genetics, National Cancer Institute, National Institutes of Health (NIH), Rockville, MD, United States.

Classifications MeSH