Clinical, Radiologic, Pathologic, and Molecular Characteristics of Long-Term Survivors of Diffuse Intrinsic Pontine Glioma (DIPG): A Collaborative Report From the International and European Society for Pediatric Oncology DIPG Registries: A Collaborative Report From the International and European Society for Pediatric Oncology DIPG Registries
Gespeichert in:
| Titel: | Clinical, Radiologic, Pathologic, and Molecular Characteristics of Long-Term Survivors of Diffuse Intrinsic Pontine Glioma (DIPG): A Collaborative Report From the International and European Society for Pediatric Oncology DIPG Registries: A Collaborative Report From the International and European Society for Pediatric Oncology DIPG Registries |
|---|---|
| Autoren: | Hetal Dholaria, Joshua J Baugh, Javad Nazarian, Torsten Pietsch, Brigitte Bison, Nancy Yanez Escorza, Maryam Fouladi, Katherine E. Warren, Lili Miles, Adam Lane, Brooklyn Chaney, André O. von Bueren, Pascale Varlet, William P. Vandertop, Sarah Leary, Christine Fuller, David T.W. Jones, Niclas Colditz, Darren Hargrave, Nathalie Boddaert, Rachid Drissi, Esther Hulleman, Nicholas K. Foreman, Marzia Giagnacovo, Chie Schin Shih, Gertjan J.L. Kaspers, Esther Sanchez, Murali Chintagumpala, Géraldine Giraud, Tim Hassall, Lindsey M. Hoffman, Elke Pfaff, Gerrit H. Gielen, Jacques Grill, Piergiorgio Modena, Eric Bouffet, Mark W. Kieran, Monika Warmuth-Metz, Raphael Calmon, Ute Bartels, James L. Leach, Nicholas G. Gottardo, Veronica Biassoni, Emmett Broxson, Stefan M. Pfister, Chris Jones, Dannis G. van Vuurden, Marion Hoffmann, Alberto Broniscer, Christof M. Kramm, Nada Jabado, Nicolas U. Gerber, Anne Sophie Carret, Martin Benesch, David Castel, Soumen Khatua, Blaise V. Jones, Michael A. Grotzer, Sophie E. M. Veldhuijzen van Zanten, Roger J. Packer, Melanie Comito, Dominik Sturm, Jane E. Minturn, Maura Massimino, Guirish A. Solanki, David S. Ziegler, Manila Antonelli, Simon Bailey, Stéphanie Puget, Filip Jadrijevic Cvrlje, Cynthia Hawkins, Renee Doughman, Stewart Goldman |
| Weitere Verfasser: | Hoffman, Lindsey M., Veldhuijzen van Zanten, Sophie E.M., Colditz, Niclas, Baugh, Joshua, Chaney, Brooklyn, Hoffmann, Marion, Lane, Adam, Fuller, Christine, Miles, Lili, Hawkins, Cynthia, Bartels, Ute, Bouffet, Eric, Goldman, Stewart, Leary, Sarah, Foreman, Nicholas K., Packer, Roger, Warren, Katherine E., Broniscer, Alberto, Kieran, Mark W., Minturn, Jane, Comito, Melanie, Broxson, Emmett, Shih, Chie-Schin, Khatua, Soumen, Chintagumpala, Murali, Carret, Anne Sophie, Escorza, Nancy Yanez, Hassall, Timothy, Ziegler, David S., Gottardo, Nicholas, Dholaria, Hetal, Doughman, Renee, Benesch, Martin, Drissi, Rachid, Nazarian, Javad, Jabado, Nada, Boddaert, Nathalie, Varlet, Pascale, Giraud, Géraldine, Castel, David, Puget, Stephanie, Jones, Chris, Hulleman, Esther, Modena, Piergiorgio, Giagnacovo, Marzia, Antonelli, Manila, Pietsch, Torsten, Gielen, Gerrit H., Jones, David T.W., Sturm, Dominik, Pfister, Stefan M., Gerber, Nicolas U., Grotzer, Michael A., Pfaff, Elke, von Bueren, André O., Hargrave, Darren, Solanki, Guirish A., Jadrijevic Cvrlje, Filip, Kaspers, Gertjan J.L., Vandertop, William P., Grill, Jacques, Bailey, Simon, Biassoni, Veronica, Massimino, Maura, Calmon, Raphaël, Sanchez, Esther, Bison, Brigitte, Warmuth-Metz, Monika, Leach, James, Jones, Blaise, van Vuurden, Dannis G., Kramm, Christof M., Fouladi, Maryam |
| Quelle: | Hoffman, L M, Veldhuijzen van Zanten, S E M, Colditz, N, Baugh, J, Chaney, B, Hoffmann, M, Lane, A, Fuller, C, Miles, L, Hawkins, C, Bartels, U, Bouffet, E, Goldman, S, Leary, S, Foreman, N K, Packer, R, Warren, K E, Broniscer, A, Kieran, M W, Minturn, J, Comito, M, Broxson, E, Shih, C-S, Khatua, S, Chintagumpala, M, Carret, A S, Escorza, N Y, Hassall, T, Ziegler, D S, Gottardo, N, Dholaria, H, Doughman, R, Benesch, M, Drissi, R, Nazarian, J, Jabado, N, Boddaert, N, Varlet, P, Giraud, G, Castel, D, Puget, S, Jones, C, Hulleman, E, Modena, P, Giagnacovo, M, Antonelli, M, Pietsch, T, Gielen, G H, Jones, D T W, Sturm, D, Pfister, S M, Gerber, N U, Grotzer, M A, Pfaff, E, von Bueren, A O, Hargrave, D, Solanki, G A, Jadrijevic Cvrlje, F, Kaspers, G J L, Vandertop, W P, Grill, J, Bailey, S, Biassoni, V, Massimino, M, Calmon, R, Sanchez, E, Bison, B, Warmuth-Metz, M, Leach, J, Jones, B, van Vuurden, D G, Kramm, C M & Fouladi, M 2018, 'Clinical, Radiologic, Pathologic, and Molecular Characteristics of Long-Term Survivors of Diffuse Intrinsic Pontine Glioma (DIPG) : A Collaborative Report From the International and European Society for Pediatric Oncology DIPG Registries', Journal of Clinical Oncology, vol. 36, no. 19, pp. 1963-1972. https://doi.org/10.1200/JCO.2017.75.9308 Journal of Clinical Oncology, Vol. 36, No 19 (2018) pp. 1963-1972 |
| Verlagsinformationen: | American Society of Clinical Oncology (ASCO), 2018. |
| Publikationsjahr: | 2018 |
| Schlagwörter: | Adult, Adolescent, Kaplan-Meier Estimate, Young Adult, 03 medical and health sciences, 0302 clinical medicine, Cancer Survivors, Brain Stem Neoplasms, Humans, Registries, Preschool, Child, 2. Zero hunger, ddc:618, Glioma/diagnosis/diagnostic imaging/genetics/therapy, Infant, Newborn, Infant, Brain Stem Neoplasms/diagnosis/diagnostic imaging/genetics/therapy, Glioma, Newborn, 3. Good health, diffuse intrinsic pontine glioma, brainstem malignancy, long-term survivors, DIPG, Child, Preschool, Cancer Survivors/statistics & numerical data |
| Beschreibung: | Purpose Diffuse intrinsic pontine glioma (DIPG) is a brainstem malignancy with a median survival of < 1 year. The International and European Society for Pediatric Oncology DIPG Registries collaborated to compare clinical, radiologic, and histomolecular characteristics between short-term survivors (STSs) and long-term survivors (LTSs). Materials and Methods Data abstracted from registry databases included patients from North America, Australia, Germany, Austria, Switzerland, the Netherlands, Italy, France, the United Kingdom, and Croatia. Results Among 1,130 pediatric and young adults with radiographically confirmed DIPG, 122 (11%) were excluded. Of the 1,008 remaining patients, 101 (10%) were LTSs (survival ≥ 2 years). Median survival time was 11 months (interquartile range, 7.5 to 16 months), and 1-, 2-, 3-, 4-, and 5-year survival rates were 42.3% (95% CI, 38.1% to 44.1%), 9.6% (95% CI, 7.8% to 11.3%), 4.3% (95% CI, 3.2% to 5.8%), 3.2% (95% CI, 2.4% to 4.6%), and 2.2% (95% CI, 1.4% to 3.4%), respectively. LTSs, compared with STSs, more commonly presented at age < 3 or > 10 years (11% v 3% and 33% v 23%, respectively; P < .001) and with longer symptom duration ( P < .001). STSs, compared with LTSs, more commonly presented with cranial nerve palsy (83% v 73%, respectively; P = .008), ring enhancement (38% v 23%, respectively; P = .007), necrosis (42% v 26%, respectively; P = .009), and extrapontine extension (92% v 86%, respectively; P = .04). LTSs more commonly received systemic therapy at diagnosis (88% v 75% for STSs; P = .005). Biopsies and autopsies were performed in 299 patients (30%) and 77 patients (10%), respectively; 181 tumors (48%) were molecularly characterized. LTSs were more likely to harbor a HIST1H3B mutation (odds ratio, 1.28; 95% CI, 1.1 to 1.5; P = .002). Conclusion We report clinical, radiologic, and molecular factors that correlate with survival in children and young adults with DIPG, which are important for risk stratification in future clinical trials. |
| Publikationsart: | Article |
| Dateibeschreibung: | application/pdf; Print-Electronic; application/vnd.openxmlformats-officedocument.spreadsheetml.sheet |
| Sprache: | English |
| ISSN: | 1527-7755 0732-183X |
| DOI: | 10.1200/jco.2017.75.9308 |
| Zugangs-URL: | https://europepmc.org/articles/pmc6075859?pdf=render https://pubmed.ncbi.nlm.nih.gov/29746225 http://www.ncbi.nlm.nih.gov/pubmed/29746225 https://europepmc.org/abstract/MED/29746225 https://archive-ouverte.unige.ch/unige:128726/ATTACHMENT01 https://ascopubs.org/doi/full/10.1200/JCO.2017.75.9308 https://www.narcis.nl/publication/RecordID/oai%3Apure.amc.nl%3Apublications%2Fbb39c06b-17e3-46d1-89b1-a03c546c1e7e https://hsrc.himmelfarb.gwu.edu/smhs_intsysbio_facpubs/249/ https://research.vumc.nl/en/publications/866f4b1d-85f7-43fe-91ca-d2ce31b3cbb6 https://archive-ouverte.unige.ch/unige:128726 https://doi.org/10.1200/jco.2017.75.9308 https://archive-ouverte.unige.ch/unige:128726 https://resolver.sub.uni-goettingen.de/purl?gro-2/77619 https://hdl.handle.net/11573/1110559 https://doi.org/10.1200/JCO.2017.75.9308 https://discovery-pp.ucl.ac.uk/id/eprint/10049511/ |
| Dokumentencode: | edsair.doi.dedup.....7fda8a6c7092120ff72e6fb9b260ba2d |
| Datenbank: | OpenAIRE |
Nájsť tento článok vo Web of Science | Abstract: | Purpose Diffuse intrinsic pontine glioma (DIPG) is a brainstem malignancy with a median survival of < 1 year. The International and European Society for Pediatric Oncology DIPG Registries collaborated to compare clinical, radiologic, and histomolecular characteristics between short-term survivors (STSs) and long-term survivors (LTSs). Materials and Methods Data abstracted from registry databases included patients from North America, Australia, Germany, Austria, Switzerland, the Netherlands, Italy, France, the United Kingdom, and Croatia. Results Among 1,130 pediatric and young adults with radiographically confirmed DIPG, 122 (11%) were excluded. Of the 1,008 remaining patients, 101 (10%) were LTSs (survival ≥ 2 years). Median survival time was 11 months (interquartile range, 7.5 to 16 months), and 1-, 2-, 3-, 4-, and 5-year survival rates were 42.3% (95% CI, 38.1% to 44.1%), 9.6% (95% CI, 7.8% to 11.3%), 4.3% (95% CI, 3.2% to 5.8%), 3.2% (95% CI, 2.4% to 4.6%), and 2.2% (95% CI, 1.4% to 3.4%), respectively. LTSs, compared with STSs, more commonly presented at age < 3 or > 10 years (11% v 3% and 33% v 23%, respectively; P < .001) and with longer symptom duration ( P < .001). STSs, compared with LTSs, more commonly presented with cranial nerve palsy (83% v 73%, respectively; P = .008), ring enhancement (38% v 23%, respectively; P = .007), necrosis (42% v 26%, respectively; P = .009), and extrapontine extension (92% v 86%, respectively; P = .04). LTSs more commonly received systemic therapy at diagnosis (88% v 75% for STSs; P = .005). Biopsies and autopsies were performed in 299 patients (30%) and 77 patients (10%), respectively; 181 tumors (48%) were molecularly characterized. LTSs were more likely to harbor a HIST1H3B mutation (odds ratio, 1.28; 95% CI, 1.1 to 1.5; P = .002). Conclusion We report clinical, radiologic, and molecular factors that correlate with survival in children and young adults with DIPG, which are important for risk stratification in future clinical trials. |
|---|---|
| ISSN: | 15277755 0732183X |
| DOI: | 10.1200/jco.2017.75.9308 |