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HGG-11. Clinical characteristics and clinical evolution of a large cohort of pediatric patients with primary central nervous system (CNS) tumors and tropomyosin receptor kinase (TRK) fusion.
BACKGROUND: TRK fusions are detected in less than 3% of CNS tumors. Given their rarity, there are limited data on the clinical course of these patients. METHODS: We contacted 166 oncology centers worldwide to retrieve data on patients with TRK fusion-driven CNS tumors. Data extracted included demogr...
| Autores principales: | , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , , |
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| Formato: | Online Artículo Texto |
| Lenguaje: | English |
| Publicado: |
Oxford University Press
2022
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| Materias: | |
| Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9164744/ http://dx.doi.org/10.1093/neuonc/noac079.226 |
| _version_ | 1784720207808299008 |
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| author | Lamoureux, Audrey-Anne Fisher, Michael Lemelle, Lauriane Pfaff, Elke Kramm, Christof De Wilde, Bram Kazanowska, Bernarda Hutter, Caroline Pfister, Stefan M Sturm, Dominik Jones, David Orbach, Daniel Pierron, Gaëlle Raskin, Scott Drilon, Alexander Diamond, Eli Harada, Guilherme Zapotocky, Michal Ellezam, Benjamin Weil, Alexander G Venne, Dominic Barritault, Marc Leblond, Pierre Coltin, Hallie Hammad, Rawan Tabori, Uri Hawkins, Cynthia Hansford, Jordan R Meyran, Deborah Erker, Craig McFadden, Kathryn Sato, Mariko Gottardo, Nicholas G Dholaria, Hetal Nørøxe, Dorte Schou Goto, Hiroaki Ziegler, David S Lin, Frank Y Parsons, Donald Williams Lindsay, Holly Wong, Tai-Tong Liu, Yen-Lin Wu, Kuo-Sheng Franson, Andrea Flynn Hwang, Eugene Aguilar-Bonilla, Ana Cheng, Sylvia Cacciotti, Chantel Massimino, Maura Schiavello, Elisabetta Wood, Paul Hoffman, Lindsey M Cappellano, Andréa Lassaletta, Alvaro Van Damme, An Llort, Anna Gerber, Nicolas U Ceruso, Mariella Spalato Bendel, Anne E Skrypek, Maggie Hamideh, Dima Mushtaq, Naureen Walter, Andrew Jabado, Nada Alsahlawi, Aysha Farmer, Jean-Pierre Abadi, Christina Coleman Mueller, Sabine Mazewski, Claire Aguilera, Dolly Robison, Nathan O’Halloran, Katrina Abbou, Samuel Berlanga, Pablo Geoerger, Birgit Øra, Ingrid Moertel, Christopher L Razis, Evangelia D Vernadou, Anastasia Doz, François Laetsch, Theodore W Perreault, Sébastien |
| author_facet | Lamoureux, Audrey-Anne Fisher, Michael Lemelle, Lauriane Pfaff, Elke Kramm, Christof De Wilde, Bram Kazanowska, Bernarda Hutter, Caroline Pfister, Stefan M Sturm, Dominik Jones, David Orbach, Daniel Pierron, Gaëlle Raskin, Scott Drilon, Alexander Diamond, Eli Harada, Guilherme Zapotocky, Michal Ellezam, Benjamin Weil, Alexander G Venne, Dominic Barritault, Marc Leblond, Pierre Coltin, Hallie Hammad, Rawan Tabori, Uri Hawkins, Cynthia Hansford, Jordan R Meyran, Deborah Erker, Craig McFadden, Kathryn Sato, Mariko Gottardo, Nicholas G Dholaria, Hetal Nørøxe, Dorte Schou Goto, Hiroaki Ziegler, David S Lin, Frank Y Parsons, Donald Williams Lindsay, Holly Wong, Tai-Tong Liu, Yen-Lin Wu, Kuo-Sheng Franson, Andrea Flynn Hwang, Eugene Aguilar-Bonilla, Ana Cheng, Sylvia Cacciotti, Chantel Massimino, Maura Schiavello, Elisabetta Wood, Paul Hoffman, Lindsey M Cappellano, Andréa Lassaletta, Alvaro Van Damme, An Llort, Anna Gerber, Nicolas U Ceruso, Mariella Spalato Bendel, Anne E Skrypek, Maggie Hamideh, Dima Mushtaq, Naureen Walter, Andrew Jabado, Nada Alsahlawi, Aysha Farmer, Jean-Pierre Abadi, Christina Coleman Mueller, Sabine Mazewski, Claire Aguilera, Dolly Robison, Nathan O’Halloran, Katrina Abbou, Samuel Berlanga, Pablo Geoerger, Birgit Øra, Ingrid Moertel, Christopher L Razis, Evangelia D Vernadou, Anastasia Doz, François Laetsch, Theodore W Perreault, Sébastien |
| author_sort | Lamoureux, Audrey-Anne |
| collection | PubMed |
| description | BACKGROUND: TRK fusions are detected in less than 3% of CNS tumors. Given their rarity, there are limited data on the clinical course of these patients. METHODS: We contacted 166 oncology centers worldwide to retrieve data on patients with TRK fusion-driven CNS tumors. Data extracted included demographics, histopathology, NTRK gene fusion, treatment modalities and outcomes. Patients less than 18 years of age at diagnosis were included in this analysis. RESULTS: Seventy-three pediatric patients with TRK fusion-driven primary CNS tumors were identified. Median age at diagnosis was 2.4 years (range 0.0–17.8) and 60.2 % were male. NTRK2 gene fusions were found in 37 patients (50.7%), NTRK1 and NTRK3 aberrations were detected in 19 (26.0%) and 17 (23.3%), respectively. Tumor types included 38 high-grade gliomas (HGG; 52.1%), 20 low-grade gliomas (LGG; 27.4%), 4 embryonal tumors (5.5%) and 11 others (15.1%). Median follow-up was 46.5 months (range 3-226). During the course of their disease, a total of 62 (84.9%) patients underwent surgery with a treatment intent, 50 (68.5%) patients received chemotherapy, 35 (47.9%) patients received radiation therapy, while 34 (46.6%) patients received NTRK inhibitors (3 as first line treatment). Twenty-four (32.9%) had no progression including 9 LGG (45%) and 9 HGG (23.6%). At last follow-up, only one (5.6%-18 evaluable) patient with LGG died compared to 11 with HGG (35.5%-31 evaluable). For LGG the median progression-free survival (PFS) after the first line of treatment was 17 months (95% CI: 0.0-35.5) and median overall survival (OS) was not reached. For patients with HGG the median PFS was 30 months (95% CI: 11.9-48.1) and median OS was 182 months (95% CI 20.2-343.8). CONCLUSIONS: We report the largest cohort of pediatric patients with TRK fusion-driven primary CNS tumors. These results will help us to better understand clinical evolution and compare outcomes with ongoing clinical trials. |
| format | Online Article Text |
| id | pubmed-9164744 |
| institution | National Center for Biotechnology Information |
| language | English |
| publishDate | 2022 |
| publisher | Oxford University Press |
| record_format | MEDLINE/PubMed |
| spelling | pubmed-91647442022-06-05 HGG-11. Clinical characteristics and clinical evolution of a large cohort of pediatric patients with primary central nervous system (CNS) tumors and tropomyosin receptor kinase (TRK) fusion. Lamoureux, Audrey-Anne Fisher, Michael Lemelle, Lauriane Pfaff, Elke Kramm, Christof De Wilde, Bram Kazanowska, Bernarda Hutter, Caroline Pfister, Stefan M Sturm, Dominik Jones, David Orbach, Daniel Pierron, Gaëlle Raskin, Scott Drilon, Alexander Diamond, Eli Harada, Guilherme Zapotocky, Michal Ellezam, Benjamin Weil, Alexander G Venne, Dominic Barritault, Marc Leblond, Pierre Coltin, Hallie Hammad, Rawan Tabori, Uri Hawkins, Cynthia Hansford, Jordan R Meyran, Deborah Erker, Craig McFadden, Kathryn Sato, Mariko Gottardo, Nicholas G Dholaria, Hetal Nørøxe, Dorte Schou Goto, Hiroaki Ziegler, David S Lin, Frank Y Parsons, Donald Williams Lindsay, Holly Wong, Tai-Tong Liu, Yen-Lin Wu, Kuo-Sheng Franson, Andrea Flynn Hwang, Eugene Aguilar-Bonilla, Ana Cheng, Sylvia Cacciotti, Chantel Massimino, Maura Schiavello, Elisabetta Wood, Paul Hoffman, Lindsey M Cappellano, Andréa Lassaletta, Alvaro Van Damme, An Llort, Anna Gerber, Nicolas U Ceruso, Mariella Spalato Bendel, Anne E Skrypek, Maggie Hamideh, Dima Mushtaq, Naureen Walter, Andrew Jabado, Nada Alsahlawi, Aysha Farmer, Jean-Pierre Abadi, Christina Coleman Mueller, Sabine Mazewski, Claire Aguilera, Dolly Robison, Nathan O’Halloran, Katrina Abbou, Samuel Berlanga, Pablo Geoerger, Birgit Øra, Ingrid Moertel, Christopher L Razis, Evangelia D Vernadou, Anastasia Doz, François Laetsch, Theodore W Perreault, Sébastien Neuro Oncol High Grade Glioma BACKGROUND: TRK fusions are detected in less than 3% of CNS tumors. Given their rarity, there are limited data on the clinical course of these patients. METHODS: We contacted 166 oncology centers worldwide to retrieve data on patients with TRK fusion-driven CNS tumors. Data extracted included demographics, histopathology, NTRK gene fusion, treatment modalities and outcomes. Patients less than 18 years of age at diagnosis were included in this analysis. RESULTS: Seventy-three pediatric patients with TRK fusion-driven primary CNS tumors were identified. Median age at diagnosis was 2.4 years (range 0.0–17.8) and 60.2 % were male. NTRK2 gene fusions were found in 37 patients (50.7%), NTRK1 and NTRK3 aberrations were detected in 19 (26.0%) and 17 (23.3%), respectively. Tumor types included 38 high-grade gliomas (HGG; 52.1%), 20 low-grade gliomas (LGG; 27.4%), 4 embryonal tumors (5.5%) and 11 others (15.1%). Median follow-up was 46.5 months (range 3-226). During the course of their disease, a total of 62 (84.9%) patients underwent surgery with a treatment intent, 50 (68.5%) patients received chemotherapy, 35 (47.9%) patients received radiation therapy, while 34 (46.6%) patients received NTRK inhibitors (3 as first line treatment). Twenty-four (32.9%) had no progression including 9 LGG (45%) and 9 HGG (23.6%). At last follow-up, only one (5.6%-18 evaluable) patient with LGG died compared to 11 with HGG (35.5%-31 evaluable). For LGG the median progression-free survival (PFS) after the first line of treatment was 17 months (95% CI: 0.0-35.5) and median overall survival (OS) was not reached. For patients with HGG the median PFS was 30 months (95% CI: 11.9-48.1) and median OS was 182 months (95% CI 20.2-343.8). CONCLUSIONS: We report the largest cohort of pediatric patients with TRK fusion-driven primary CNS tumors. These results will help us to better understand clinical evolution and compare outcomes with ongoing clinical trials. Oxford University Press 2022-06-03 /pmc/articles/PMC9164744/ http://dx.doi.org/10.1093/neuonc/noac079.226 Text en © The Author(s) 2022. Published by Oxford University Press on behalf of the Society for Neuro-Oncology. https://creativecommons.org/licenses/by-nc/4.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial License (https://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com |
| spellingShingle | High Grade Glioma Lamoureux, Audrey-Anne Fisher, Michael Lemelle, Lauriane Pfaff, Elke Kramm, Christof De Wilde, Bram Kazanowska, Bernarda Hutter, Caroline Pfister, Stefan M Sturm, Dominik Jones, David Orbach, Daniel Pierron, Gaëlle Raskin, Scott Drilon, Alexander Diamond, Eli Harada, Guilherme Zapotocky, Michal Ellezam, Benjamin Weil, Alexander G Venne, Dominic Barritault, Marc Leblond, Pierre Coltin, Hallie Hammad, Rawan Tabori, Uri Hawkins, Cynthia Hansford, Jordan R Meyran, Deborah Erker, Craig McFadden, Kathryn Sato, Mariko Gottardo, Nicholas G Dholaria, Hetal Nørøxe, Dorte Schou Goto, Hiroaki Ziegler, David S Lin, Frank Y Parsons, Donald Williams Lindsay, Holly Wong, Tai-Tong Liu, Yen-Lin Wu, Kuo-Sheng Franson, Andrea Flynn Hwang, Eugene Aguilar-Bonilla, Ana Cheng, Sylvia Cacciotti, Chantel Massimino, Maura Schiavello, Elisabetta Wood, Paul Hoffman, Lindsey M Cappellano, Andréa Lassaletta, Alvaro Van Damme, An Llort, Anna Gerber, Nicolas U Ceruso, Mariella Spalato Bendel, Anne E Skrypek, Maggie Hamideh, Dima Mushtaq, Naureen Walter, Andrew Jabado, Nada Alsahlawi, Aysha Farmer, Jean-Pierre Abadi, Christina Coleman Mueller, Sabine Mazewski, Claire Aguilera, Dolly Robison, Nathan O’Halloran, Katrina Abbou, Samuel Berlanga, Pablo Geoerger, Birgit Øra, Ingrid Moertel, Christopher L Razis, Evangelia D Vernadou, Anastasia Doz, François Laetsch, Theodore W Perreault, Sébastien HGG-11. Clinical characteristics and clinical evolution of a large cohort of pediatric patients with primary central nervous system (CNS) tumors and tropomyosin receptor kinase (TRK) fusion. |
| title | HGG-11. Clinical characteristics and clinical evolution of a large cohort of pediatric patients with primary central nervous system (CNS) tumors and tropomyosin receptor kinase (TRK) fusion. |
| title_full | HGG-11. Clinical characteristics and clinical evolution of a large cohort of pediatric patients with primary central nervous system (CNS) tumors and tropomyosin receptor kinase (TRK) fusion. |
| title_fullStr | HGG-11. Clinical characteristics and clinical evolution of a large cohort of pediatric patients with primary central nervous system (CNS) tumors and tropomyosin receptor kinase (TRK) fusion. |
| title_full_unstemmed | HGG-11. Clinical characteristics and clinical evolution of a large cohort of pediatric patients with primary central nervous system (CNS) tumors and tropomyosin receptor kinase (TRK) fusion. |
| title_short | HGG-11. Clinical characteristics and clinical evolution of a large cohort of pediatric patients with primary central nervous system (CNS) tumors and tropomyosin receptor kinase (TRK) fusion. |
| title_sort | hgg-11. clinical characteristics and clinical evolution of a large cohort of pediatric patients with primary central nervous system (cns) tumors and tropomyosin receptor kinase (trk) fusion. |
| topic | High Grade Glioma |
| url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9164744/ http://dx.doi.org/10.1093/neuonc/noac079.226 |
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