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dc.contributor.authorDineen, Robert A.
dc.contributor.authorMorgan, Paul S.
dc.contributor.authorJaspan, Timothy
dc.date.accessioned2024-02-07T11:37:46Z
dc.date.available2024-02-07T11:37:46Z
dc.date.issued2022
dc.identifier.citationRodriguez, D., Calmon, R., Aliaga, E.S., Warren, D., Warmuth-Metz, M., Jones, C., Mackay, A., Varlet, P., Le Deley, M., Hargrave, D., Canete, A., Massimino, M., Azizi, A.A., Saran, F., Zahlmann, G., Garcia, J., Vassal, G., Grill, J., Peet, A., Dineen, R.A., Morgan, P.S. and Jaspan, T. (2022) 'MRI and molecular characterization of pediatric high-grade midline thalamic gliomas: The HERBY phase II trial', Radiology, 304(1), pp. 174-182. doi: 10.1148/radiol.211464 https://doi.org/10.1148/radiol.211464.en_US
dc.identifier.issn1527-1315
dc.identifier.issn0033-8419
dc.identifier.urihttp://hdl.handle.net/20.500.12904/18215
dc.descriptionAvailable to read on the publisher's website here: https://doi.org/10.1148/radiol.211464.en_US
dc.description.abstractBackground Diffuse midline gliomas (DMG) are characterized by a high incidence of H3 K27 mutations and poorer outcome. The HERBY trial has provided one of the largest cohorts of pediatric DMGs with available radiologic, histologic-genotypic, and survival data. Purpose To define MRI and molecular characteristics of DMG. Materials and Methods This study is a secondary analysis of a prospective trial (HERBY; ClinicalTrials.gov identifier, NCT01390948) undertaken between October 2011 and February 2016. Among 121 HERBY participants, 50 had midline nonpontine-based tumors. Midline high-grade gliomas were reclassified into DMG H3 K27 mutant, H3 wild type with enhancer of zest homologs inhibitory protein overexpression, epidermal growth factor receptormutant, or not otherwise stated. The epicenter of each tumor and other radiologic characteristics were ascertained from MRI and correlated with the new subtype classification, histopathologic characteristics, surgical extent, and outcome parameters. Kaplan-Meier curves and log-rank tests were applied to determine and describe survival differences between groups. Results There were 42 participants (mean age, 12 years +/- 4 [SD]; 23 girls) with radiologically evaluable thalamic-based DMG. Eighteen had partial thalamic involvement (12 thalamopulvinar, six anteromedial), 10 involved a whole thalamus, nine had unithalamic tumors with diffuse contiguous extension, and five had bithalamic tumors (two symmetric, three partial). Twenty-eight participants had DMG H3 K27 mutant tumors; there were no differences in outcome compared with other DMGs (n = 4). Participants who underwent major debulking or total or near-total resection had longer overall survival (OS): 18.5 months vs 11.4 months (P = .02). Enrolled participants who developed leptomeningeal metastatic dissemination before starting treatment had worse outcomes (event-free survival, 2.9 months vs 8.0 months [P = .02]; OS, 11.4 months vs 18.5 months [P = .004]). Conclusion Thalamic involvement of diffuse midline gliomas ranged from localized partial thalamic to holo- or bithalamic with diffuse contiguous spread and had poor outcomes, irrespective of H3 K27 subtype alterations. Leptomeningeal dissemination and less than 50% surgical resection were adverse risk factors for survival. Clinical trial registration no. NCT01390948 © RSNA, 2022 Online supplemental material is available for this article. See also the editorial by Widjaja in this issue.
dc.description.urihttps://doi.org/10.1148/radiol.211464en_US
dc.language.isoenen_US
dc.subjectGliomaen_US
dc.subjectMagnetic resonance imagingen_US
dc.subjectPaediatricsen_US
dc.titleMRI and molecular characterization of pediatric high-grade midline thalamic gliomas: The HERBY phase II trialen_US
dc.typeArticleen_US
rioxxterms.funderDefault funderen_US
rioxxterms.identifier.projectDefault projecten_US
rioxxterms.versionVoRen_US
rioxxterms.versionofrecord10.1148/radiol.211464en_US
rioxxterms.typeJournal Article/Reviewen_US
refterms.dateFCD2024-02-07T11:37:48Z
refterms.versionFCDVoR
refterms.panelUnspecifieden_US
html.description.abstractBackground Diffuse midline gliomas (DMG) are characterized by a high incidence of H3 K27 mutations and poorer outcome. The HERBY trial has provided one of the largest cohorts of pediatric DMGs with available radiologic, histologic-genotypic, and survival data. Purpose To define MRI and molecular characteristics of DMG. Materials and Methods This study is a secondary analysis of a prospective trial (HERBY; ClinicalTrials.gov identifier, NCT01390948) undertaken between October 2011 and February 2016. Among 121 HERBY participants, 50 had midline nonpontine-based tumors. Midline high-grade gliomas were reclassified into DMG H3 K27 mutant, H3 wild type with enhancer of zest homologs inhibitory protein overexpression, epidermal growth factor receptormutant, or not otherwise stated. The epicenter of each tumor and other radiologic characteristics were ascertained from MRI and correlated with the new subtype classification, histopathologic characteristics, surgical extent, and outcome parameters. Kaplan-Meier curves and log-rank tests were applied to determine and describe survival differences between groups. Results There were 42 participants (mean age, 12 years +/- 4 [SD]; 23 girls) with radiologically evaluable thalamic-based DMG. Eighteen had partial thalamic involvement (12 thalamopulvinar, six anteromedial), 10 involved a whole thalamus, nine had unithalamic tumors with diffuse contiguous extension, and five had bithalamic tumors (two symmetric, three partial). Twenty-eight participants had DMG H3 K27 mutant tumors; there were no differences in outcome compared with other DMGs (n = 4). Participants who underwent major debulking or total or near-total resection had longer overall survival (OS): 18.5 months vs 11.4 months (P = .02). Enrolled participants who developed leptomeningeal metastatic dissemination before starting treatment had worse outcomes (event-free survival, 2.9 months vs 8.0 months [P = .02]; OS, 11.4 months vs 18.5 months [P = .004]). Conclusion Thalamic involvement of diffuse midline gliomas ranged from localized partial thalamic to holo- or bithalamic with diffuse contiguous spread and had poor outcomes, irrespective of H3 K27 subtype alterations. Leptomeningeal dissemination and less than 50% surgical resection were adverse risk factors for survival. Clinical trial registration no. NCT01390948 © RSNA, 2022 Online supplemental material is available for this article. See also the editorial by Widjaja in this issue.en_US
rioxxterms.funder.project94a427429a5bcfef7dd04c33360d80cden_US


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