Adult H3K27M mutated thalamic glioma patients display a better prognosis than unmutated patients

Background Adult thalamic gliomas are a rare entity whose management is challenging for physicians. The aim of this study is to describe the characteristics and prognostic factors of thalamic gliomas in adult patients. Methods We retrospectively analyzed the clinical, neuro-radiological, histologica...

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Veröffentlicht in:Journal of neuro-oncology 2022-02, Vol.156 (3), p.615-623
Hauptverfasser: Grimaldi, Stéphan, Harlay, Vincent, Appay, Romain, Bequet, Céline, Petrirena, Grégorio, Campello, Chantal, Barrié, Maryline, Autran, Didier, Boissonneau, Sébastien, Graillon, Thomas, Figarella-Branger, Dominique, Nanni, Isabelle, Chinot, Olivier, Tabouret, Emeline
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container_issue 3
container_start_page 615
container_title Journal of neuro-oncology
container_volume 156
creator Grimaldi, Stéphan
Harlay, Vincent
Appay, Romain
Bequet, Céline
Petrirena, Grégorio
Campello, Chantal
Barrié, Maryline
Autran, Didier
Boissonneau, Sébastien
Graillon, Thomas
Figarella-Branger, Dominique
Nanni, Isabelle
Chinot, Olivier
Tabouret, Emeline
description Background Adult thalamic gliomas are a rare entity whose management is challenging for physicians. The aim of this study is to describe the characteristics and prognostic factors of thalamic gliomas in adult patients. Methods We retrospectively analyzed the clinical, neuro-radiological, histological, and molecular characteristics of all cases of adult thalamic glioma in our regional center. Results We included 38 adult patients. Median age at diagnosis was 56.5 years old (range, 24–80). Median KPS at diagnosis was 70%. Two-thirds of patients presented with tumor necrosis on MRI. Bithalamic lesions were present in four patients. The median volume of enhancement associated with lesions was relatively small (14 mm 3 ). Two patients had undergone partial surgical resection. All other patients underwent biopsy. Median PFS was 7.1 months (95% CI [3.7–10.5]) and median OS was 15.6 months (95% CI [11.7–19.6]). Among 20 patients with available tumor samples for molecular analyses, only 4 (20%) presented with H3K27M mutation. Patients with H3K27M mutation had longer survival compared to those without. Finally, we identified a long-term survivor population characterized by a younger age, no cognitive impairment, low steroid dose treatment and the presence of H3K27M mutation. Conclusion Thalamic adult glioma differs from bithalamic glioma in children with regards to its clinical, radiological and molecular profiles. Long-term survival is observed in young patients with limited symptoms and H3K27M mutation. A larger prospective cohort is needed to validate these findings.
doi_str_mv 10.1007/s11060-022-03943-7
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The aim of this study is to describe the characteristics and prognostic factors of thalamic gliomas in adult patients. Methods We retrospectively analyzed the clinical, neuro-radiological, histological, and molecular characteristics of all cases of adult thalamic glioma in our regional center. Results We included 38 adult patients. Median age at diagnosis was 56.5 years old (range, 24–80). Median KPS at diagnosis was 70%. Two-thirds of patients presented with tumor necrosis on MRI. Bithalamic lesions were present in four patients. The median volume of enhancement associated with lesions was relatively small (14 mm 3 ). Two patients had undergone partial surgical resection. All other patients underwent biopsy. Median PFS was 7.1 months (95% CI [3.7–10.5]) and median OS was 15.6 months (95% CI [11.7–19.6]). Among 20 patients with available tumor samples for molecular analyses, only 4 (20%) presented with H3K27M mutation. Patients with H3K27M mutation had longer survival compared to those without. Finally, we identified a long-term survivor population characterized by a younger age, no cognitive impairment, low steroid dose treatment and the presence of H3K27M mutation. Conclusion Thalamic adult glioma differs from bithalamic glioma in children with regards to its clinical, radiological and molecular profiles. Long-term survival is observed in young patients with limited symptoms and H3K27M mutation. A larger prospective cohort is needed to validate these findings.</description><identifier>ISSN: 0167-594X</identifier><identifier>EISSN: 1573-7373</identifier><identifier>DOI: 10.1007/s11060-022-03943-7</identifier><identifier>PMID: 34994963</identifier><language>eng</language><publisher>New York: Springer US</publisher><subject>Adult ; Biopsy ; Brain Neoplasms - genetics ; Brain Neoplasms - therapy ; Brain tumors ; Clinical Study ; Cognitive ability ; Diagnosis ; Glioma ; Glioma - genetics ; Glioma - therapy ; Histones - genetics ; Humans ; Life Sciences ; Magnetic resonance imaging ; Medical prognosis ; Medicine ; Medicine &amp; Public Health ; Mutation ; Neurology ; Oncology ; Patients ; Prognosis ; Retrospective Studies ; Survival ; Thalamus ; Thalamus - pathology</subject><ispartof>Journal of neuro-oncology, 2022-02, Vol.156 (3), p.615-623</ispartof><rights>The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature 2022</rights><rights>2022. The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.</rights><rights>The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature 2022.</rights><rights>Distributed under a Creative Commons Attribution 4.0 International License</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c409t-5e89ea515233d32364066d95ceabea6e4d45776241d80e06a5faef155b1b3de33</citedby><cites>FETCH-LOGICAL-c409t-5e89ea515233d32364066d95ceabea6e4d45776241d80e06a5faef155b1b3de33</cites><orcidid>0000-0002-4031-2178</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktopdf>$$Uhttps://link.springer.com/content/pdf/10.1007/s11060-022-03943-7$$EPDF$$P50$$Gspringer$$H</linktopdf><linktohtml>$$Uhttps://link.springer.com/10.1007/s11060-022-03943-7$$EHTML$$P50$$Gspringer$$H</linktohtml><link.rule.ids>230,314,780,784,885,27924,27925,41488,42557,51319</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/34994963$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink><backlink>$$Uhttps://hal.science/hal-03976537$$DView record in HAL$$Hfree_for_read</backlink></links><search><creatorcontrib>Grimaldi, Stéphan</creatorcontrib><creatorcontrib>Harlay, Vincent</creatorcontrib><creatorcontrib>Appay, Romain</creatorcontrib><creatorcontrib>Bequet, Céline</creatorcontrib><creatorcontrib>Petrirena, Grégorio</creatorcontrib><creatorcontrib>Campello, Chantal</creatorcontrib><creatorcontrib>Barrié, Maryline</creatorcontrib><creatorcontrib>Autran, Didier</creatorcontrib><creatorcontrib>Boissonneau, Sébastien</creatorcontrib><creatorcontrib>Graillon, Thomas</creatorcontrib><creatorcontrib>Figarella-Branger, Dominique</creatorcontrib><creatorcontrib>Nanni, Isabelle</creatorcontrib><creatorcontrib>Chinot, Olivier</creatorcontrib><creatorcontrib>Tabouret, Emeline</creatorcontrib><title>Adult H3K27M mutated thalamic glioma patients display a better prognosis than unmutated patients</title><title>Journal of neuro-oncology</title><addtitle>J Neurooncol</addtitle><addtitle>J Neurooncol</addtitle><description>Background Adult thalamic gliomas are a rare entity whose management is challenging for physicians. The aim of this study is to describe the characteristics and prognostic factors of thalamic gliomas in adult patients. Methods We retrospectively analyzed the clinical, neuro-radiological, histological, and molecular characteristics of all cases of adult thalamic glioma in our regional center. Results We included 38 adult patients. Median age at diagnosis was 56.5 years old (range, 24–80). Median KPS at diagnosis was 70%. Two-thirds of patients presented with tumor necrosis on MRI. Bithalamic lesions were present in four patients. The median volume of enhancement associated with lesions was relatively small (14 mm 3 ). Two patients had undergone partial surgical resection. All other patients underwent biopsy. Median PFS was 7.1 months (95% CI [3.7–10.5]) and median OS was 15.6 months (95% CI [11.7–19.6]). Among 20 patients with available tumor samples for molecular analyses, only 4 (20%) presented with H3K27M mutation. Patients with H3K27M mutation had longer survival compared to those without. Finally, we identified a long-term survivor population characterized by a younger age, no cognitive impairment, low steroid dose treatment and the presence of H3K27M mutation. Conclusion Thalamic adult glioma differs from bithalamic glioma in children with regards to its clinical, radiological and molecular profiles. Long-term survival is observed in young patients with limited symptoms and H3K27M mutation. 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The aim of this study is to describe the characteristics and prognostic factors of thalamic gliomas in adult patients. Methods We retrospectively analyzed the clinical, neuro-radiological, histological, and molecular characteristics of all cases of adult thalamic glioma in our regional center. Results We included 38 adult patients. Median age at diagnosis was 56.5 years old (range, 24–80). Median KPS at diagnosis was 70%. Two-thirds of patients presented with tumor necrosis on MRI. Bithalamic lesions were present in four patients. The median volume of enhancement associated with lesions was relatively small (14 mm 3 ). Two patients had undergone partial surgical resection. All other patients underwent biopsy. Median PFS was 7.1 months (95% CI [3.7–10.5]) and median OS was 15.6 months (95% CI [11.7–19.6]). Among 20 patients with available tumor samples for molecular analyses, only 4 (20%) presented with H3K27M mutation. Patients with H3K27M mutation had longer survival compared to those without. Finally, we identified a long-term survivor population characterized by a younger age, no cognitive impairment, low steroid dose treatment and the presence of H3K27M mutation. Conclusion Thalamic adult glioma differs from bithalamic glioma in children with regards to its clinical, radiological and molecular profiles. Long-term survival is observed in young patients with limited symptoms and H3K27M mutation. A larger prospective cohort is needed to validate these findings.</abstract><cop>New York</cop><pub>Springer US</pub><pmid>34994963</pmid><doi>10.1007/s11060-022-03943-7</doi><tpages>9</tpages><orcidid>https://orcid.org/0000-0002-4031-2178</orcidid></addata></record>
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subjects Adult
Biopsy
Brain Neoplasms - genetics
Brain Neoplasms - therapy
Brain tumors
Clinical Study
Cognitive ability
Diagnosis
Glioma
Glioma - genetics
Glioma - therapy
Histones - genetics
Humans
Life Sciences
Magnetic resonance imaging
Medical prognosis
Medicine
Medicine & Public Health
Mutation
Neurology
Oncology
Patients
Prognosis
Retrospective Studies
Survival
Thalamus
Thalamus - pathology
title Adult H3K27M mutated thalamic glioma patients display a better prognosis than unmutated patients
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