High-grade gliomas in adolescents and young adults highlight histomolecular differences from their adult and pediatric counterparts
Abstract Background Considering that pediatric high-grade gliomas (HGGs) are biologically distinct from their adult counterparts, the objective of this study was to define the landscape of HGGs in adolescents and young adults (AYAs). Methods We performed a multicentric retrospective study of 112 AYA...
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| Veröffentlicht in: | Neuro-oncology (Charlottesville, Va.) Va.), 2020-08, Vol.22 (8), p.1190-1202 |
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| creator | Roux, Alexandre Pallud, Johan Saffroy, Raphaël Edjlali-Goujon, Myriam Debily, Marie-Anne Boddaert, Nathalie Sanson, Marc Puget, Stéphanie Knafo, Steven Adam, Clovis Faillot, Thierry Cazals-Hatem, Dominique Mandonnet, Emmanuel Polivka, Marc Dorfmüller, Georges Dauta, Aurélie Desplanques, Mathilde Gareton, Albane Pages, Mélanie Tauziede-Espariat, Arnault Grill, Jacques Bourdeaut, Franck Doz, François Dhermain, Frédéric Mokhtari, Karima Chretien, Fabrice Figarella-Branger, Dominique Varlet, Pascale |
| description | Abstract
Background
Considering that pediatric high-grade gliomas (HGGs) are biologically distinct from their adult counterparts, the objective of this study was to define the landscape of HGGs in adolescents and young adults (AYAs).
Methods
We performed a multicentric retrospective study of 112 AYAs from adult and pediatric Ile-de-France neurosurgical units, treated between 1998 and 2013 to analyze their clinicoradiological and histomolecular profiles. The inclusion criteria were age between 15 and 25 years, histopathological HGG diagnosis, available clinical data, and preoperative and follow-up MRI. MRI and tumoral samples were centrally reviewed. Immunohistochemistry and complementary molecular techniques such as targeted/next-generation sequencing, whole exome sequencing, and DNA-methylation analyses were performed to achieve an integrated diagnosis according to the 2016 World Health Organization (WHO) classification.
Results
Based on 80 documented AYA patients, HGGs constitute heterogeneous clinicopathological and molecular groups, with a predominant representation of pediatric subtypes (histone H3-mutants, 40%) but also adult subtypes (isocitrate dehydrogenase [IDH] mutants, 28%) characterized by the rarity of oligodendrogliomas, IDH mutants, and 1p/19q codeletion and the relative high frequency of “rare adult IDH mutations” (20%). H3G34-mutants (14%) represent the most specific subgroup in AYAs. In the H3K27-mutant subgroup, non-brainstem diffuse midline gliomas are more frequent (66.7%) than diffuse intrinsic pontine gliomas (23.8%), contrary to what is observed in children. We found that WHO grade has no prognostic value, but molecular subgrouping has major prognostic importance.
Conclusions
HGGs in AYAs could benefit from a specific classification, driven by molecular subtyping rather than age group. Collaborative efforts are needed from pediatric and adult neuro-oncology teams to improve the management of HGGs in AYAs. |
| doi_str_mv | 10.1093/neuonc/noaa024 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_pubme</sourceid><recordid>TN_cdi_pubmedcentral_primary_oai_pubmedcentral_nih_gov_7594566</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><oup_id>10.1093/neuonc/noaa024</oup_id><sourcerecordid>2352052200</sourcerecordid><originalsourceid>FETCH-LOGICAL-c458t-e46e50f7f7cf95850825d8d49327c422cfef539e06f2dac116dab991023f19343</originalsourceid><addsrcrecordid>eNqFkb1v1TAUxS0EoqWwMqKMdEjrjziJF6SqojykJ7HAbLn2dWLk2MFOKnXuP45f8ygfC4Plq-tzfvb1QegtwRcEC3YZYI1BX4aoFKbNM3RKOGU179v2-WNN656T7gS9yvk7xpTwlrxEJ4xiyjvan6KHnRvGekjKQDV4FyeVKxcqZaKHrCEsuVLBVPdxDUPprr40xmLxZS2lykucilSvXqXKOGshQdCQK5viVC0juLTZHjEzGKeW5HSlC3CBNKu05NfohVU-w5vjfoa-3Xz8er2r918-fb6-2te64f1SQ9MCx7aznbaC9xz3lJveNILRTjeUaguWMwG4tdQoTUhr1K0QBFNmiWANO0MfNu683k5gDtMl5eWc3KTSvYzKyb9PghvlEO9kx0XD27YAzjfA-I9td7WXhx5mGIueNXekaN8fL0vxxwp5kZMrH-q9ChDXLCnjFJd8MC7Si02qU8w5gX1iEywPKcstZXlMuRje_TnIk_xXrL8fGtf5f7CfB763Uw</addsrcrecordid><sourcetype>Open Access Repository</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2352052200</pqid></control><display><type>article</type><title>High-grade gliomas in adolescents and young adults highlight histomolecular differences from their adult and pediatric counterparts</title><source>Oxford University Press Journals All Titles (1996-Current)</source><source>MEDLINE</source><source>Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals</source><source>PubMed Central</source><creator>Roux, Alexandre ; Pallud, Johan ; Saffroy, Raphaël ; Edjlali-Goujon, Myriam ; Debily, Marie-Anne ; Boddaert, Nathalie ; Sanson, Marc ; Puget, Stéphanie ; Knafo, Steven ; Adam, Clovis ; Faillot, Thierry ; Cazals-Hatem, Dominique ; Mandonnet, Emmanuel ; Polivka, Marc ; Dorfmüller, Georges ; Dauta, Aurélie ; Desplanques, Mathilde ; Gareton, Albane ; Pages, Mélanie ; Tauziede-Espariat, Arnault ; Grill, Jacques ; Bourdeaut, Franck ; Doz, François ; Dhermain, Frédéric ; Mokhtari, Karima ; Chretien, Fabrice ; Figarella-Branger, Dominique ; Varlet, Pascale</creator><creatorcontrib>Roux, Alexandre ; Pallud, Johan ; Saffroy, Raphaël ; Edjlali-Goujon, Myriam ; Debily, Marie-Anne ; Boddaert, Nathalie ; Sanson, Marc ; Puget, Stéphanie ; Knafo, Steven ; Adam, Clovis ; Faillot, Thierry ; Cazals-Hatem, Dominique ; Mandonnet, Emmanuel ; Polivka, Marc ; Dorfmüller, Georges ; Dauta, Aurélie ; Desplanques, Mathilde ; Gareton, Albane ; Pages, Mélanie ; Tauziede-Espariat, Arnault ; Grill, Jacques ; Bourdeaut, Franck ; Doz, François ; Dhermain, Frédéric ; Mokhtari, Karima ; Chretien, Fabrice ; Figarella-Branger, Dominique ; Varlet, Pascale</creatorcontrib><description>Abstract
Background
Considering that pediatric high-grade gliomas (HGGs) are biologically distinct from their adult counterparts, the objective of this study was to define the landscape of HGGs in adolescents and young adults (AYAs).
Methods
We performed a multicentric retrospective study of 112 AYAs from adult and pediatric Ile-de-France neurosurgical units, treated between 1998 and 2013 to analyze their clinicoradiological and histomolecular profiles. The inclusion criteria were age between 15 and 25 years, histopathological HGG diagnosis, available clinical data, and preoperative and follow-up MRI. MRI and tumoral samples were centrally reviewed. Immunohistochemistry and complementary molecular techniques such as targeted/next-generation sequencing, whole exome sequencing, and DNA-methylation analyses were performed to achieve an integrated diagnosis according to the 2016 World Health Organization (WHO) classification.
Results
Based on 80 documented AYA patients, HGGs constitute heterogeneous clinicopathological and molecular groups, with a predominant representation of pediatric subtypes (histone H3-mutants, 40%) but also adult subtypes (isocitrate dehydrogenase [IDH] mutants, 28%) characterized by the rarity of oligodendrogliomas, IDH mutants, and 1p/19q codeletion and the relative high frequency of “rare adult IDH mutations” (20%). H3G34-mutants (14%) represent the most specific subgroup in AYAs. In the H3K27-mutant subgroup, non-brainstem diffuse midline gliomas are more frequent (66.7%) than diffuse intrinsic pontine gliomas (23.8%), contrary to what is observed in children. We found that WHO grade has no prognostic value, but molecular subgrouping has major prognostic importance.
Conclusions
HGGs in AYAs could benefit from a specific classification, driven by molecular subtyping rather than age group. Collaborative efforts are needed from pediatric and adult neuro-oncology teams to improve the management of HGGs in AYAs.</description><identifier>ISSN: 1522-8517</identifier><identifier>EISSN: 1523-5866</identifier><identifier>DOI: 10.1093/neuonc/noaa024</identifier><identifier>PMID: 32025728</identifier><language>eng</language><publisher>US: Oxford University Press</publisher><subject>Adolescent ; Adult ; Age Factors ; Biochemistry, Molecular Biology ; Brain Neoplasms - genetics ; Brain Neoplasms - pathology ; Cancer ; Female ; Genomics ; Glioma - enzymology ; Glioma - genetics ; Glioma - pathology ; Human health and pathology ; Humans ; Isocitrate Dehydrogenase - genetics ; Life Sciences ; Male ; Mutation ; Neoplasm Grading ; Neurobiology ; Neurons and Cognition ; Oligodendroglioma - enzymology ; Oligodendroglioma - genetics ; Oligodendroglioma - pathology ; Pediatric Neuro-Oncology ; Retrospective Studies ; Tissues and Organs ; Young Adult</subject><ispartof>Neuro-oncology (Charlottesville, Va.), 2020-08, Vol.22 (8), p.1190-1202</ispartof><rights>The Author(s) 2020. Published by Oxford University Press on behalf of the Society for Neuro-Oncology. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com 2020</rights><rights>The Author(s) 2020. Published by Oxford University Press on behalf of the Society for Neuro-Oncology. All rights reserved. For permissions, please e-mail: journals.permissions@oup.com.</rights><rights>Distributed under a Creative Commons Attribution 4.0 International License</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c458t-e46e50f7f7cf95850825d8d49327c422cfef539e06f2dac116dab991023f19343</citedby><cites>FETCH-LOGICAL-c458t-e46e50f7f7cf95850825d8d49327c422cfef539e06f2dac116dab991023f19343</cites><orcidid>0000-0002-1652-9844 ; 0000-0002-3650-8581 ; 0000-0003-0991-7774 ; 0000-0002-3604-887X ; 0000-0002-8164-1517 ; 0000-0002-1813-8476 ; 0000-0003-0230-9282</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC7594566/pdf/$$EPDF$$P50$$Gpubmedcentral$$H</linktopdf><linktohtml>$$Uhttps://www.ncbi.nlm.nih.gov/pmc/articles/PMC7594566/$$EHTML$$P50$$Gpubmedcentral$$H</linktohtml><link.rule.ids>230,314,723,776,780,881,1578,27901,27902,53766,53768</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/32025728$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink><backlink>$$Uhttps://hal.science/hal-03009834$$DView record in HAL$$Hfree_for_read</backlink></links><search><creatorcontrib>Roux, Alexandre</creatorcontrib><creatorcontrib>Pallud, Johan</creatorcontrib><creatorcontrib>Saffroy, Raphaël</creatorcontrib><creatorcontrib>Edjlali-Goujon, Myriam</creatorcontrib><creatorcontrib>Debily, Marie-Anne</creatorcontrib><creatorcontrib>Boddaert, Nathalie</creatorcontrib><creatorcontrib>Sanson, Marc</creatorcontrib><creatorcontrib>Puget, Stéphanie</creatorcontrib><creatorcontrib>Knafo, Steven</creatorcontrib><creatorcontrib>Adam, Clovis</creatorcontrib><creatorcontrib>Faillot, Thierry</creatorcontrib><creatorcontrib>Cazals-Hatem, Dominique</creatorcontrib><creatorcontrib>Mandonnet, Emmanuel</creatorcontrib><creatorcontrib>Polivka, Marc</creatorcontrib><creatorcontrib>Dorfmüller, Georges</creatorcontrib><creatorcontrib>Dauta, Aurélie</creatorcontrib><creatorcontrib>Desplanques, Mathilde</creatorcontrib><creatorcontrib>Gareton, Albane</creatorcontrib><creatorcontrib>Pages, Mélanie</creatorcontrib><creatorcontrib>Tauziede-Espariat, Arnault</creatorcontrib><creatorcontrib>Grill, Jacques</creatorcontrib><creatorcontrib>Bourdeaut, Franck</creatorcontrib><creatorcontrib>Doz, François</creatorcontrib><creatorcontrib>Dhermain, Frédéric</creatorcontrib><creatorcontrib>Mokhtari, Karima</creatorcontrib><creatorcontrib>Chretien, Fabrice</creatorcontrib><creatorcontrib>Figarella-Branger, Dominique</creatorcontrib><creatorcontrib>Varlet, Pascale</creatorcontrib><title>High-grade gliomas in adolescents and young adults highlight histomolecular differences from their adult and pediatric counterparts</title><title>Neuro-oncology (Charlottesville, Va.)</title><addtitle>Neuro Oncol</addtitle><description>Abstract
Background
Considering that pediatric high-grade gliomas (HGGs) are biologically distinct from their adult counterparts, the objective of this study was to define the landscape of HGGs in adolescents and young adults (AYAs).
Methods
We performed a multicentric retrospective study of 112 AYAs from adult and pediatric Ile-de-France neurosurgical units, treated between 1998 and 2013 to analyze their clinicoradiological and histomolecular profiles. The inclusion criteria were age between 15 and 25 years, histopathological HGG diagnosis, available clinical data, and preoperative and follow-up MRI. MRI and tumoral samples were centrally reviewed. Immunohistochemistry and complementary molecular techniques such as targeted/next-generation sequencing, whole exome sequencing, and DNA-methylation analyses were performed to achieve an integrated diagnosis according to the 2016 World Health Organization (WHO) classification.
Results
Based on 80 documented AYA patients, HGGs constitute heterogeneous clinicopathological and molecular groups, with a predominant representation of pediatric subtypes (histone H3-mutants, 40%) but also adult subtypes (isocitrate dehydrogenase [IDH] mutants, 28%) characterized by the rarity of oligodendrogliomas, IDH mutants, and 1p/19q codeletion and the relative high frequency of “rare adult IDH mutations” (20%). H3G34-mutants (14%) represent the most specific subgroup in AYAs. In the H3K27-mutant subgroup, non-brainstem diffuse midline gliomas are more frequent (66.7%) than diffuse intrinsic pontine gliomas (23.8%), contrary to what is observed in children. We found that WHO grade has no prognostic value, but molecular subgrouping has major prognostic importance.
Conclusions
HGGs in AYAs could benefit from a specific classification, driven by molecular subtyping rather than age group. Collaborative efforts are needed from pediatric and adult neuro-oncology teams to improve the management of HGGs in AYAs.</description><subject>Adolescent</subject><subject>Adult</subject><subject>Age Factors</subject><subject>Biochemistry, Molecular Biology</subject><subject>Brain Neoplasms - genetics</subject><subject>Brain Neoplasms - pathology</subject><subject>Cancer</subject><subject>Female</subject><subject>Genomics</subject><subject>Glioma - enzymology</subject><subject>Glioma - genetics</subject><subject>Glioma - pathology</subject><subject>Human health and pathology</subject><subject>Humans</subject><subject>Isocitrate Dehydrogenase - genetics</subject><subject>Life Sciences</subject><subject>Male</subject><subject>Mutation</subject><subject>Neoplasm Grading</subject><subject>Neurobiology</subject><subject>Neurons and Cognition</subject><subject>Oligodendroglioma - enzymology</subject><subject>Oligodendroglioma - genetics</subject><subject>Oligodendroglioma - pathology</subject><subject>Pediatric Neuro-Oncology</subject><subject>Retrospective Studies</subject><subject>Tissues and Organs</subject><subject>Young Adult</subject><issn>1522-8517</issn><issn>1523-5866</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2020</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqFkb1v1TAUxS0EoqWwMqKMdEjrjziJF6SqojykJ7HAbLn2dWLk2MFOKnXuP45f8ygfC4Plq-tzfvb1QegtwRcEC3YZYI1BX4aoFKbNM3RKOGU179v2-WNN656T7gS9yvk7xpTwlrxEJ4xiyjvan6KHnRvGekjKQDV4FyeVKxcqZaKHrCEsuVLBVPdxDUPprr40xmLxZS2lykucilSvXqXKOGshQdCQK5viVC0juLTZHjEzGKeW5HSlC3CBNKu05NfohVU-w5vjfoa-3Xz8er2r918-fb6-2te64f1SQ9MCx7aznbaC9xz3lJveNILRTjeUaguWMwG4tdQoTUhr1K0QBFNmiWANO0MfNu683k5gDtMl5eWc3KTSvYzKyb9PghvlEO9kx0XD27YAzjfA-I9td7WXhx5mGIueNXekaN8fL0vxxwp5kZMrH-q9ChDXLCnjFJd8MC7Si02qU8w5gX1iEywPKcstZXlMuRje_TnIk_xXrL8fGtf5f7CfB763Uw</recordid><startdate>20200817</startdate><enddate>20200817</enddate><creator>Roux, Alexandre</creator><creator>Pallud, Johan</creator><creator>Saffroy, Raphaël</creator><creator>Edjlali-Goujon, Myriam</creator><creator>Debily, Marie-Anne</creator><creator>Boddaert, Nathalie</creator><creator>Sanson, Marc</creator><creator>Puget, Stéphanie</creator><creator>Knafo, Steven</creator><creator>Adam, Clovis</creator><creator>Faillot, Thierry</creator><creator>Cazals-Hatem, Dominique</creator><creator>Mandonnet, Emmanuel</creator><creator>Polivka, Marc</creator><creator>Dorfmüller, Georges</creator><creator>Dauta, Aurélie</creator><creator>Desplanques, Mathilde</creator><creator>Gareton, Albane</creator><creator>Pages, Mélanie</creator><creator>Tauziede-Espariat, Arnault</creator><creator>Grill, Jacques</creator><creator>Bourdeaut, Franck</creator><creator>Doz, François</creator><creator>Dhermain, Frédéric</creator><creator>Mokhtari, Karima</creator><creator>Chretien, Fabrice</creator><creator>Figarella-Branger, Dominique</creator><creator>Varlet, Pascale</creator><general>Oxford University Press</general><general>Oxford University Press (OUP)</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope><scope>1XC</scope><scope>5PM</scope><orcidid>https://orcid.org/0000-0002-1652-9844</orcidid><orcidid>https://orcid.org/0000-0002-3650-8581</orcidid><orcidid>https://orcid.org/0000-0003-0991-7774</orcidid><orcidid>https://orcid.org/0000-0002-3604-887X</orcidid><orcidid>https://orcid.org/0000-0002-8164-1517</orcidid><orcidid>https://orcid.org/0000-0002-1813-8476</orcidid><orcidid>https://orcid.org/0000-0003-0230-9282</orcidid></search><sort><creationdate>20200817</creationdate><title>High-grade gliomas in adolescents and young adults highlight histomolecular differences from their adult and pediatric counterparts</title><author>Roux, Alexandre ; Pallud, Johan ; Saffroy, Raphaël ; Edjlali-Goujon, Myriam ; Debily, Marie-Anne ; Boddaert, Nathalie ; Sanson, Marc ; Puget, Stéphanie ; Knafo, Steven ; Adam, Clovis ; Faillot, Thierry ; Cazals-Hatem, Dominique ; Mandonnet, Emmanuel ; Polivka, Marc ; Dorfmüller, Georges ; Dauta, Aurélie ; Desplanques, Mathilde ; Gareton, Albane ; Pages, Mélanie ; Tauziede-Espariat, Arnault ; Grill, Jacques ; Bourdeaut, Franck ; Doz, François ; Dhermain, Frédéric ; Mokhtari, Karima ; Chretien, Fabrice ; Figarella-Branger, Dominique ; Varlet, Pascale</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c458t-e46e50f7f7cf95850825d8d49327c422cfef539e06f2dac116dab991023f19343</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2020</creationdate><topic>Adolescent</topic><topic>Adult</topic><topic>Age Factors</topic><topic>Biochemistry, Molecular Biology</topic><topic>Brain Neoplasms - genetics</topic><topic>Brain Neoplasms - pathology</topic><topic>Cancer</topic><topic>Female</topic><topic>Genomics</topic><topic>Glioma - enzymology</topic><topic>Glioma - genetics</topic><topic>Glioma - pathology</topic><topic>Human health and pathology</topic><topic>Humans</topic><topic>Isocitrate Dehydrogenase - genetics</topic><topic>Life Sciences</topic><topic>Male</topic><topic>Mutation</topic><topic>Neoplasm Grading</topic><topic>Neurobiology</topic><topic>Neurons and Cognition</topic><topic>Oligodendroglioma - enzymology</topic><topic>Oligodendroglioma - genetics</topic><topic>Oligodendroglioma - pathology</topic><topic>Pediatric Neuro-Oncology</topic><topic>Retrospective Studies</topic><topic>Tissues and Organs</topic><topic>Young Adult</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Roux, Alexandre</creatorcontrib><creatorcontrib>Pallud, Johan</creatorcontrib><creatorcontrib>Saffroy, Raphaël</creatorcontrib><creatorcontrib>Edjlali-Goujon, Myriam</creatorcontrib><creatorcontrib>Debily, Marie-Anne</creatorcontrib><creatorcontrib>Boddaert, Nathalie</creatorcontrib><creatorcontrib>Sanson, Marc</creatorcontrib><creatorcontrib>Puget, Stéphanie</creatorcontrib><creatorcontrib>Knafo, Steven</creatorcontrib><creatorcontrib>Adam, Clovis</creatorcontrib><creatorcontrib>Faillot, Thierry</creatorcontrib><creatorcontrib>Cazals-Hatem, Dominique</creatorcontrib><creatorcontrib>Mandonnet, Emmanuel</creatorcontrib><creatorcontrib>Polivka, Marc</creatorcontrib><creatorcontrib>Dorfmüller, Georges</creatorcontrib><creatorcontrib>Dauta, Aurélie</creatorcontrib><creatorcontrib>Desplanques, Mathilde</creatorcontrib><creatorcontrib>Gareton, Albane</creatorcontrib><creatorcontrib>Pages, Mélanie</creatorcontrib><creatorcontrib>Tauziede-Espariat, Arnault</creatorcontrib><creatorcontrib>Grill, Jacques</creatorcontrib><creatorcontrib>Bourdeaut, Franck</creatorcontrib><creatorcontrib>Doz, François</creatorcontrib><creatorcontrib>Dhermain, Frédéric</creatorcontrib><creatorcontrib>Mokhtari, Karima</creatorcontrib><creatorcontrib>Chretien, Fabrice</creatorcontrib><creatorcontrib>Figarella-Branger, Dominique</creatorcontrib><creatorcontrib>Varlet, Pascale</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><collection>Hyper Article en Ligne (HAL)</collection><collection>PubMed Central (Full Participant titles)</collection><jtitle>Neuro-oncology (Charlottesville, Va.)</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Roux, Alexandre</au><au>Pallud, Johan</au><au>Saffroy, Raphaël</au><au>Edjlali-Goujon, Myriam</au><au>Debily, Marie-Anne</au><au>Boddaert, Nathalie</au><au>Sanson, Marc</au><au>Puget, Stéphanie</au><au>Knafo, Steven</au><au>Adam, Clovis</au><au>Faillot, Thierry</au><au>Cazals-Hatem, Dominique</au><au>Mandonnet, Emmanuel</au><au>Polivka, Marc</au><au>Dorfmüller, Georges</au><au>Dauta, Aurélie</au><au>Desplanques, Mathilde</au><au>Gareton, Albane</au><au>Pages, Mélanie</au><au>Tauziede-Espariat, Arnault</au><au>Grill, Jacques</au><au>Bourdeaut, Franck</au><au>Doz, François</au><au>Dhermain, Frédéric</au><au>Mokhtari, Karima</au><au>Chretien, Fabrice</au><au>Figarella-Branger, Dominique</au><au>Varlet, Pascale</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>High-grade gliomas in adolescents and young adults highlight histomolecular differences from their adult and pediatric counterparts</atitle><jtitle>Neuro-oncology (Charlottesville, Va.)</jtitle><addtitle>Neuro Oncol</addtitle><date>2020-08-17</date><risdate>2020</risdate><volume>22</volume><issue>8</issue><spage>1190</spage><epage>1202</epage><pages>1190-1202</pages><issn>1522-8517</issn><eissn>1523-5866</eissn><abstract>Abstract
Background
Considering that pediatric high-grade gliomas (HGGs) are biologically distinct from their adult counterparts, the objective of this study was to define the landscape of HGGs in adolescents and young adults (AYAs).
Methods
We performed a multicentric retrospective study of 112 AYAs from adult and pediatric Ile-de-France neurosurgical units, treated between 1998 and 2013 to analyze their clinicoradiological and histomolecular profiles. The inclusion criteria were age between 15 and 25 years, histopathological HGG diagnosis, available clinical data, and preoperative and follow-up MRI. MRI and tumoral samples were centrally reviewed. Immunohistochemistry and complementary molecular techniques such as targeted/next-generation sequencing, whole exome sequencing, and DNA-methylation analyses were performed to achieve an integrated diagnosis according to the 2016 World Health Organization (WHO) classification.
Results
Based on 80 documented AYA patients, HGGs constitute heterogeneous clinicopathological and molecular groups, with a predominant representation of pediatric subtypes (histone H3-mutants, 40%) but also adult subtypes (isocitrate dehydrogenase [IDH] mutants, 28%) characterized by the rarity of oligodendrogliomas, IDH mutants, and 1p/19q codeletion and the relative high frequency of “rare adult IDH mutations” (20%). H3G34-mutants (14%) represent the most specific subgroup in AYAs. In the H3K27-mutant subgroup, non-brainstem diffuse midline gliomas are more frequent (66.7%) than diffuse intrinsic pontine gliomas (23.8%), contrary to what is observed in children. We found that WHO grade has no prognostic value, but molecular subgrouping has major prognostic importance.
Conclusions
HGGs in AYAs could benefit from a specific classification, driven by molecular subtyping rather than age group. Collaborative efforts are needed from pediatric and adult neuro-oncology teams to improve the management of HGGs in AYAs.</abstract><cop>US</cop><pub>Oxford University Press</pub><pmid>32025728</pmid><doi>10.1093/neuonc/noaa024</doi><tpages>13</tpages><orcidid>https://orcid.org/0000-0002-1652-9844</orcidid><orcidid>https://orcid.org/0000-0002-3650-8581</orcidid><orcidid>https://orcid.org/0000-0003-0991-7774</orcidid><orcidid>https://orcid.org/0000-0002-3604-887X</orcidid><orcidid>https://orcid.org/0000-0002-8164-1517</orcidid><orcidid>https://orcid.org/0000-0002-1813-8476</orcidid><orcidid>https://orcid.org/0000-0003-0230-9282</orcidid><oa>free_for_read</oa></addata></record> |
| fulltext | fulltext |
| identifier | ISSN: 1522-8517 |
| ispartof | Neuro-oncology (Charlottesville, Va.), 2020-08, Vol.22 (8), p.1190-1202 |
| issn | 1522-8517 1523-5866 |
| language | eng |
| recordid | cdi_pubmedcentral_primary_oai_pubmedcentral_nih_gov_7594566 |
| source | Oxford University Press Journals All Titles (1996-Current); MEDLINE; Elektronische Zeitschriftenbibliothek - Frei zugängliche E-Journals; PubMed Central |
| subjects | Adolescent Adult Age Factors Biochemistry, Molecular Biology Brain Neoplasms - genetics Brain Neoplasms - pathology Cancer Female Genomics Glioma - enzymology Glioma - genetics Glioma - pathology Human health and pathology Humans Isocitrate Dehydrogenase - genetics Life Sciences Male Mutation Neoplasm Grading Neurobiology Neurons and Cognition Oligodendroglioma - enzymology Oligodendroglioma - genetics Oligodendroglioma - pathology Pediatric Neuro-Oncology Retrospective Studies Tissues and Organs Young Adult |
| title | High-grade gliomas in adolescents and young adults highlight histomolecular differences from their adult and pediatric counterparts |
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