Prognostic Implication of Patient Age in H3K27M-Mutant Midline Gliomas

IntroductionPediatric and adult H3K27M-mutant midline gliomas have variable clinical presentations, prognoses, and molecular backgrounds. In this study, we integrated data from published studies to investigate the differences between these two groups.MethodsPubMed and Web of Science were searched fo...

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Main Authors: Huy Gia Vuong, Tam N. M. Ngo, Hieu Trong Le, Andrew Jea, Maya Hrachova, James Battiste, Rene McNall-Knapp, Ian F. Dunn
Format: Article
Language:English
Published: Frontiers Media S.A. 2022-03-01
Series:Frontiers in Oncology
Subjects:
Online Access:https://www.frontiersin.org/articles/10.3389/fonc.2022.858148/full
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author Huy Gia Vuong
Tam N. M. Ngo
Hieu Trong Le
Andrew Jea
Maya Hrachova
James Battiste
Rene McNall-Knapp
Ian F. Dunn
author_facet Huy Gia Vuong
Tam N. M. Ngo
Hieu Trong Le
Andrew Jea
Maya Hrachova
James Battiste
Rene McNall-Knapp
Ian F. Dunn
author_sort Huy Gia Vuong
collection DOAJ
description IntroductionPediatric and adult H3K27M-mutant midline gliomas have variable clinical presentations, prognoses, and molecular backgrounds. In this study, we integrated data from published studies to investigate the differences between these two groups.MethodsPubMed and Web of Science were searched for potential data. Studies were included if they had available individual participant data on patients age of H3K27M-mutant midline gliomas. For time-to-event analyses, Kaplan-Meier analysis and Cox regression models were carried out; corresponding hazard ratios (HR) and 95% confidence intervals (CI) were computed to analyze the impact of age and clinical covariates on progression-free survival (PFS) and overall survival (OS).ResultsWe included 43 studies comprising 272 adults and 657 pediatric midline gliomas with H3K27M mutation for analyses. In adults, there was a male predilection whereas females were slightly more common than males in the pediatric group. Spinal cord tumors were more frequent in adults. The prevalence of H3.1 K27M mutation was significantly higher in the pediatric cohort. Compared to adult patients, pediatric H3K27M-mutant midline gliomas exhibited more aggressive features including higher rates of pathologic features of high-grade tumors and Ki67 proliferation index, and had a shorter PFS and OS. Genetically, ACVR1 mutations were more common whereas MGMT methylation, FGFR1, and NF1 mutations were less prevalent in the pediatric cohort.ConclusionPediatric H3K27M-mutant midline gliomas were demographically, clinically, and molecularly distinct from adult patients, highlighting an opportunity to refine the risk stratification for these neoplasms.
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spelling doaj.art-4a012ac2bf844f458ebddf41c0ecf3d92022-12-22T00:04:50ZengFrontiers Media S.A.Frontiers in Oncology2234-943X2022-03-011210.3389/fonc.2022.858148858148Prognostic Implication of Patient Age in H3K27M-Mutant Midline GliomasHuy Gia Vuong0Tam N. M. Ngo1Hieu Trong Le2Andrew Jea3Maya Hrachova4James Battiste5Rene McNall-Knapp6Ian F. Dunn7Department of Neurosurgery, The University of Oklahoma Health Sciences Center, Oklahoma University, Oklahoma City, OK, United StatesFaculty of Medicine, Pham Ngoc Thach University of Medicine, Ho Chi Minh City, VietnamDepartment of Pathology, University of Medicine and Pharmacy at Ho Chi Minh City, Ho Chi Minh City, VietnamDepartment of Neurosurgery, Division of Pediatric Neurosurgery, Oklahoma Children’s Hospital, The University of Oklahoma Health Sciences Center, Oklahoma University, Oklahoma City, OK, United StatesDepartment of Neurosurgery, The University of Oklahoma Health Sciences Center, Oklahoma University, Oklahoma City, OK, United StatesDepartment of Neurosurgery, The University of Oklahoma Health Sciences Center, Oklahoma University, Oklahoma City, OK, United StatesDepartment of Pediatrics, The University of Oklahoma Health Sciences Center, Oklahoma University, Oklahoma City, OK, United StatesDepartment of Neurosurgery, The University of Oklahoma Health Sciences Center, Oklahoma University, Oklahoma City, OK, United StatesIntroductionPediatric and adult H3K27M-mutant midline gliomas have variable clinical presentations, prognoses, and molecular backgrounds. In this study, we integrated data from published studies to investigate the differences between these two groups.MethodsPubMed and Web of Science were searched for potential data. Studies were included if they had available individual participant data on patients age of H3K27M-mutant midline gliomas. For time-to-event analyses, Kaplan-Meier analysis and Cox regression models were carried out; corresponding hazard ratios (HR) and 95% confidence intervals (CI) were computed to analyze the impact of age and clinical covariates on progression-free survival (PFS) and overall survival (OS).ResultsWe included 43 studies comprising 272 adults and 657 pediatric midline gliomas with H3K27M mutation for analyses. In adults, there was a male predilection whereas females were slightly more common than males in the pediatric group. Spinal cord tumors were more frequent in adults. The prevalence of H3.1 K27M mutation was significantly higher in the pediatric cohort. Compared to adult patients, pediatric H3K27M-mutant midline gliomas exhibited more aggressive features including higher rates of pathologic features of high-grade tumors and Ki67 proliferation index, and had a shorter PFS and OS. Genetically, ACVR1 mutations were more common whereas MGMT methylation, FGFR1, and NF1 mutations were less prevalent in the pediatric cohort.ConclusionPediatric H3K27M-mutant midline gliomas were demographically, clinically, and molecularly distinct from adult patients, highlighting an opportunity to refine the risk stratification for these neoplasms.https://www.frontiersin.org/articles/10.3389/fonc.2022.858148/fullH3K27Mmidline gliomaH3F3AHIST1H3B/Cpediatricadult
spellingShingle Huy Gia Vuong
Tam N. M. Ngo
Hieu Trong Le
Andrew Jea
Maya Hrachova
James Battiste
Rene McNall-Knapp
Ian F. Dunn
Prognostic Implication of Patient Age in H3K27M-Mutant Midline Gliomas
Frontiers in Oncology
H3K27M
midline glioma
H3F3A
HIST1H3B/C
pediatric
adult
title Prognostic Implication of Patient Age in H3K27M-Mutant Midline Gliomas
title_full Prognostic Implication of Patient Age in H3K27M-Mutant Midline Gliomas
title_fullStr Prognostic Implication of Patient Age in H3K27M-Mutant Midline Gliomas
title_full_unstemmed Prognostic Implication of Patient Age in H3K27M-Mutant Midline Gliomas
title_short Prognostic Implication of Patient Age in H3K27M-Mutant Midline Gliomas
title_sort prognostic implication of patient age in h3k27m mutant midline gliomas
topic H3K27M
midline glioma
H3F3A
HIST1H3B/C
pediatric
adult
url https://www.frontiersin.org/articles/10.3389/fonc.2022.858148/full
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