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The pediatric precision oncology INFORM registry: clinical outcome and benefit for patients with very high-evidence targets

INFORM is a prospective, multi-national registry gathering clinical and molecular data of relapsed, progressive or high-risk pediatric cancer patients. This report describes long-term follow-up of 519 patients in whom molecular alterations were evaluated according to a pre-defined 7-scale target pri...

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Hauptverfasser: Tilburg, Cornelis M. van (VerfasserIn) , Pfaff, Elke (VerfasserIn) , Pajtler, Kristian Wilfried (VerfasserIn) , Langenberg, Karin (VerfasserIn) , Fiesel, Petra (VerfasserIn) , Jones, Barbara Christine (VerfasserIn) , Balasubramanian, Gnana Prakash (VerfasserIn) , Stark, Sebastian (VerfasserIn) , Johann, Pascal-David (VerfasserIn) , Blattner-Johnson, Mirjam (VerfasserIn) , Schramm, Kathrin (VerfasserIn) , Dikow, Nicola (VerfasserIn) , Hirsch, Steffen (VerfasserIn) , Sutter, Christian (VerfasserIn) , Grund, Kerstin (VerfasserIn) , Stackelberg, Arend von (VerfasserIn) , Kulozik, Andreas (VerfasserIn) , Lissat, Andrej (VerfasserIn) , Borkhardt, Arndt (VerfasserIn) , Meisel, Roland (VerfasserIn) , Reinhardt, Dirk (VerfasserIn) , Klusmann, Jan-Henning (VerfasserIn) , Fleischhack, Gudrun (VerfasserIn) , Tippelt, Stephan (VerfasserIn) , Schweinitz, Dietrich von (VerfasserIn) , Schmid, Irene (VerfasserIn) , Kramm, Christof M. (VerfasserIn) , Bueren, André von (VerfasserIn) , Calaminus, Gabriele (VerfasserIn) , Vorwerk, Peter (VerfasserIn) , Graf, Norbert (VerfasserIn) , Westermann, Frank (VerfasserIn) , Fischer, Matthias (VerfasserIn) , Eggert, Angelika (VerfasserIn) , Burkhardt, Birgit (VerfasserIn) , Wößmann, Wilhelm (VerfasserIn) , Nathrath, Michaela (VerfasserIn) , Hecker-Nolting, Stefanie (VerfasserIn) , Frühwald, Michael (VerfasserIn) , Schneider, Dominik T. (VerfasserIn) , Brecht, Ines B. (VerfasserIn) , Ketteler, Petra (VerfasserIn) , Fulda, Simone (VerfasserIn) , Koscielniak, Ewa (VerfasserIn) , Meister, Michael (VerfasserIn) , Scheer, Monika (VerfasserIn) , Hettmer, Simone (VerfasserIn) , Schwab, Matthias (VerfasserIn) , Tremmel, Roman (VerfasserIn) , Øra, Ingrid (VerfasserIn) , Hutter, Caroline (VerfasserIn) , Gerber, Nicolas U. (VerfasserIn) , Lohi, Olli (VerfasserIn) , Kazanowska, Bernarda (VerfasserIn) , Kattamis, Antonis (VerfasserIn) , Filippidou, Maria (VerfasserIn) , Goemans, Bianca (VerfasserIn) , Zwaan, C. Michel (VerfasserIn) , Milde, Till (VerfasserIn) , Jäger, Natalie (VerfasserIn) , Wolf, Stephan (VerfasserIn) , Reuss, David (VerfasserIn) , Sahm, Felix (VerfasserIn) , Deimling, Andreas von (VerfasserIn) , Dirksen, Uta (VerfasserIn) , Freitag, Angelika (VerfasserIn) , Witt, Ruth (VerfasserIn) , Lichter, Peter (VerfasserIn) , Kopp-Schneider, Annette (VerfasserIn) , Jones, David T. W. (VerfasserIn) , Molenaar, Jan J. (VerfasserIn) , Capper, David (VerfasserIn) , Pfister, Stefan (VerfasserIn) , Witt, Olaf (VerfasserIn)
Dokumenttyp: Article (Journal)
Sprache:Englisch
Veröffentlicht: August 9, 2021
In: Cancer discovery
Year: 2021, Jahrgang: 11, Heft: 11, Pages: 2764-2779
ISSN:2159-8290
DOI:10.1158/2159-8290.CD-21-0094
Online-Zugang:Resolving-System, lizenzpflichtig, Volltext: https://doi.org/10.1158/2159-8290.CD-21-0094
Verlag, lizenzpflichtig, Volltext: https://cancerdiscovery.aacrjournals.org/content/early/2021/07/29/2159-8290.CD-21-0094
Volltext
Verfasserangaben:Cornelis M. van Tilburg, Elke Pfaff, Kristian W. Pajtler, Karin P. S. Langenberg, Petra Fiesel, Barbara C. Jones, Gnana Prakash Balasubramanian, Sebastian Stark, Pascal D. Johann, Mirjam Blattner-Johnson, Kathrin Schramm, Nicola Dikow, Steffen Hirsch, Christian Sutter, Kerstin Grund, Arend von Stackelberg, Andreas E. Kulozik, Andrej Lissat, Arndt Borkhardt, Roland Meisel, Dirk Reinhardt, Jan-Henning Klusmann, Gudrun Fleischhack, Stephan Tippelt, Dietrich von Schweinitz, Irene Schmid, Christof M. Kramm, Andre O. von Bueren, Gabriele Calaminus, Peter Vorwerk, Norbert Graf, Frank Westermann, Matthias Fischer, Angelika Eggert, Birgit Burkhardt, Wilhelm Wossmann, Michaela Nathrath, Stefanie Hecker-Nolting, Michael C. Fruhwald, Dominik T. Schneider, Ines B. Brecht, Petra Ketteler, Simone Fulda, Ewa Koscielniak, Michael T. Meister, Monika Scheer, Simone Hettmer, Matthias Schwab, Roman Tremmel, Ingrid Ora, Caroline Hutter, Nicolas U. Gerber, Olli Lohi, Bernarda Kazanowska, Antonis Kattamis, Maria Filippidou, Bianca Goemans, C. Michel Zwaan, Till Milde, Natalie Jager, Stephan Wolf, David Reuss, Felix Sahm, Andreas von Deimling, Uta Dirksen, Angelika Freitag, Ruth Witt, Peter Lichter, Annette Kopp-Schneider, David T.W. Jones, Jan J. Molenaar, David Capper, Stefan M. Pfister, Olaf Witt

MARC

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245 1 4 |a The pediatric precision oncology INFORM registry  |b clinical outcome and benefit for patients with very high-evidence targets  |c Cornelis M. van Tilburg, Elke Pfaff, Kristian W. Pajtler, Karin P. S. Langenberg, Petra Fiesel, Barbara C. Jones, Gnana Prakash Balasubramanian, Sebastian Stark, Pascal D. Johann, Mirjam Blattner-Johnson, Kathrin Schramm, Nicola Dikow, Steffen Hirsch, Christian Sutter, Kerstin Grund, Arend von Stackelberg, Andreas E. Kulozik, Andrej Lissat, Arndt Borkhardt, Roland Meisel, Dirk Reinhardt, Jan-Henning Klusmann, Gudrun Fleischhack, Stephan Tippelt, Dietrich von Schweinitz, Irene Schmid, Christof M. Kramm, Andre O. von Bueren, Gabriele Calaminus, Peter Vorwerk, Norbert Graf, Frank Westermann, Matthias Fischer, Angelika Eggert, Birgit Burkhardt, Wilhelm Wossmann, Michaela Nathrath, Stefanie Hecker-Nolting, Michael C. Fruhwald, Dominik T. Schneider, Ines B. Brecht, Petra Ketteler, Simone Fulda, Ewa Koscielniak, Michael T. Meister, Monika Scheer, Simone Hettmer, Matthias Schwab, Roman Tremmel, Ingrid Ora, Caroline Hutter, Nicolas U. Gerber, Olli Lohi, Bernarda Kazanowska, Antonis Kattamis, Maria Filippidou, Bianca Goemans, C. Michel Zwaan, Till Milde, Natalie Jager, Stephan Wolf, David Reuss, Felix Sahm, Andreas von Deimling, Uta Dirksen, Angelika Freitag, Ruth Witt, Peter Lichter, Annette Kopp-Schneider, David T.W. Jones, Jan J. Molenaar, David Capper, Stefan M. Pfister, Olaf Witt 
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520 |a INFORM is a prospective, multi-national registry gathering clinical and molecular data of relapsed, progressive or high-risk pediatric cancer patients. This report describes long-term follow-up of 519 patients in whom molecular alterations were evaluated according to a pre-defined 7-scale target prioritization algorithm. Mean turnaround time from sample receipt to report was 25.4 days. The highest target priority level was observed in 42 patients (8.1%). Of these, twenty patients received matched targeted treatment with a median PFS of 204 days (95% CI 99 - N.A.), compared with 117 days (95% CI 106 - 143; P=0.011) in all other patients. The respective molecular targets were shown to be predictive for matched treatment response and not prognostic surrogates for improved outcome. Hereditary cancer predisposition syndromes were identified in 7.5% of patients, half of which were newly identified through the study. Integrated molecular analyses resulted in a change or refinement of diagnoses in 8.2% of cases. 
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998 |g 118119990  |a Westermann, Frank  |m 118119990:Westermann, Frank  |p 32 
998 |g 1025736567  |a Kulozik, Andreas  |m 1025736567:Kulozik, Andreas  |d 910000  |d 910500  |e 910000PK1025736567  |e 910500PK1025736567  |k 0/910000/  |k 1/910000/910500/  |p 17 
998 |g 1015236219  |a Grund, Kerstin  |m 1015236219:Grund, Kerstin  |d 910000  |d 911500  |e 910000PG1015236219  |e 911500PG1015236219  |k 0/910000/  |k 1/910000/911500/  |p 15 
998 |g 1067529160  |a Sutter, Christian  |m 1067529160:Sutter, Christian  |d 910000  |d 911500  |e 910000PS1067529160  |e 911500PS1067529160  |k 0/910000/  |k 1/910000/911500/  |p 14 
998 |g 1204283125  |a Hirsch, Steffen  |m 1204283125:Hirsch, Steffen  |d 910000  |d 911500  |e 910000PH1204283125  |e 911500PH1204283125  |k 0/910000/  |k 1/910000/911500/  |p 13 
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998 |g 1017717044  |a Johann, Pascal-David  |m 1017717044:Johann, Pascal-David  |p 9 
998 |g 134202066  |a Stark, Sebastian  |m 134202066:Stark, Sebastian  |d 910000  |d 910500  |e 910000PS134202066  |e 910500PS134202066  |k 0/910000/  |k 1/910000/910500/  |p 8 
998 |g 1078243352  |a Jones, Barbara Christine  |m 1078243352:Jones, Barbara Christine  |d 910000  |d 910500  |e 910000PJ1078243352  |e 910500PJ1078243352  |k 0/910000/  |k 1/910000/910500/  |p 6 
998 |g 1012877264  |a Pajtler, Kristian Wilfried  |m 1012877264:Pajtler, Kristian Wilfried  |d 910000  |d 910500  |e 910000PP1012877264  |e 910500PP1012877264  |k 0/910000/  |k 1/910000/910500/  |p 3 
998 |g 1037262174  |a Pfaff, Elke  |m 1037262174:Pfaff, Elke  |d 910000  |d 910500  |e 910000PP1037262174  |e 910500PP1037262174  |k 0/910000/  |k 1/910000/910500/  |p 2 
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999 |a KXP-PPN1766003079  |e 3964357839 
BIB |a Y 
SER |a journal 
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