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3. Diagnostic evaluation of paediatric autoimmune lymphoproliferative immunodeficiencies (ALPID): a prospective cohort study

Author

Abinun, Mario, Abrahamsen, Tore G., Albert, Michael H., Almalky, Mohamed, Altaf, Sadaf, Babayeva, Royala, Bakhtiar, Shahrzad, Baris, Safa, Baumann, Ulrich, Becker, Martina, Beier, Rita, Berger, Thomas, Biebl, Ariane, Bielack, Stefan S., Biskup, Saskia, Bode, Sebastian FN, Borchers, Regine, Boztug, Kaan, Brockmann, Knut, Bruwier, Annelyse, Buchholz, Bernd, Caballero-Oteyza, Andres, Cant, Andrew J., Castro, Carla N., Classen, Carl F., Claviez, Alexander, Crazzolara, Roman, Cuntz, Franziska, Dąbrowska-Leonik, Nel, Derichs, Ute, Dückers, Gregor, Eberl, Wolfgang, Ebetsberger-Dachs, Georg, Erlacher, Miriam, Fabre, Alexandre, Faletti, Laura, Farmand, Susan, Figueiredo, Antonio E., Fischer, Marco, Flaadt, Tim, Full, Hermann, Gambineri, Eleonora, Girschick, Hermann, Goldacker, Sigune, Grimbacher, Bodo, Groß, Miriam, Gruhn, Bernd, Haberfellner, Florian, Hague, Rosie, Hauch, Holger, Hauck, Fabian, Heine, Sabine, Huisman, Elise J., Jakovljevic, Gordana, James, Beki, Janda, Ales, Jones, Neil, Kaiser-Labusch, Petra, Kentouche, Karim, Knight, Julian C., Knirsch, Stephanie, Kontny, Udo, Körholz, Julia, Krenn, Thomas, Kuehnle, Ingrid, Kühne, Thomas, Lee-Dimroth, Jae-Yun, Lehmann, Hartwig, Leipold, Alfred, Meinhardt, Andrea, Minkov, Milen, Mönkemöller, Kirsten, Morbach, Henner, Mücke, Urs, Nathrath, Michaela, Naumann-Bartsch, Nora, Neth, Olaf, Niemeyer, Charlotte M., Olbrich, Peter, Ostró, Róbert, Owens, Stephen, Pac, Malgorzata, Pachlopnik Schmid, Jana, Page, Matthew J.T., Pekrun, Arnulf, Prader, Seraina, Proietti, Michele, Rajacic, Nada, Rothoeft, Tobias, Ryan, Clodagh, Salou, Sarah, Salzer, Elisabeth, Savic, Sinisa, Schilling, Freimut H., Schönberger, Stefan, Schuetz, Catharina, Schuez-Havupalo, Linnea, Schulte, Björn, Schulz, Ansgar, Schuster, Volker, Seidel, Markus G., Siepermann, Kathrin, Skomska-Pawliszak, Malgorzata, Smisek, Petr, Soomann, Maarja, Stiefel, Martina, Storck, Simone, Strahm, Brigitte, Streiter, Monika, Teltschik, Heiko-Manuel, Thalhammer, Julian, Tippelt, Stephan, Toskov, Vasil, Trück, Johannes, Vieth, Simon, von Bismarck, Philipp, Wegehaupt, Oliver, Wiesel, Thomas, Wittkowski, Helmut, Yalcin Gungoren, Ezgi, Hägele, Pauline, Staus, Paulina, Scheible, Raphael, Uhlmann, Annette, Heeg, Maximilian, Klemann, Christian, Maccari, Maria Elena, Ritterbusch, Henrike, Armstrong, Martin, Cutcutache, Ioana, Elliott, Katherine S, von Bernuth, Horst, Leahy, Timothy Ronan, Leyh, Jörg, Holzinger, Dirk, Lehmberg, Kai, Svec, Peter, Masjosthusmann, Katja, Hambleton, Sophie, Jakob, Marcus, Sparber-Sauer, Monika, Kager, Leo, Puzik, Alexander, Wolkewitz, Martin, Lorenz, Myriam Ricarda, Schwarz, Klaus, Speckmann, Carsten, Rensing-Ehl, Anne, and Ehl, Stephan

Published
2024
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4. European standard clinical practice recommendations for children and adolescents with primary and recurrent osteosarcoma

Author

van Ewijk, Roelof, Herold, Nikolas, Baecklund, Fredrik, Baumhoer, Daniel, Boye, Kjetil, Gaspar, Nathalie, Harrabi, Semi B., Haveman, Lianne M., Hecker-Nolting, Stefanie, Hiemcke-Jiwa, Laura, Martin, Valentine, Fernández, Cristina Mata, Palmerini, Emanuela, van de Sande, Michiel A.J., Strauss, Sandra J., Bielack, Stefan S., and Kager, Leo

Published
2023
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7. Osteosarcoma Arising as a Secondary Malignancy following Treatment for Hematologic Cancer: A Report of 33 Affected Patients from the Cooperative Osteosarcoma Study Group (COSS)

Author

Bielack, Stefan S., primary, Mettmann, Vanessa, additional, Baumhoer, Daniel, additional, Blattmann, Claudia, additional, Burkhardt, Birgit, additional, Deinzer, Christoph K. W., additional, Kager, Leo, additional, Kevric, Matthias, additional, Mauz-Körholz, Christine, additional, Müller-Abt, Peter, additional, Reinhardt, Dirk, additional, Sabo, Alexandru-Anton, additional, Schrappe, Martin, additional, Sorg, Benjamin, additional, Windhager, Reinhard, additional, and Hecker-Nolting, Stefanie, additional

Published
2024
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8. Osteosarcoma and causes of death: A report of 1520 deceased patients from the Cooperative Osteosarcoma Study Group (COSS)

Author

Bielack, Stefan S., Blattmann, Claudia, Borkhardt, Arndt, Csóka, Monika, Hassenpflug, Wolf, Kabíčková, Edita, Kager, Leo, Kessler, Thorsten, Kratz, Christian, Kühne, Thomas, Kevric, Matthias, Lehrnbecher, Thomas, Mayer-Steinacker, Regine, Mettmann, Vanessa, Metzler, Markus, Reichardt, Peter, Rossig, Claudia, Sorg, Benjamin, von Luettichau, Irene, Windhager, Reinhard, and Hecker-Nolting, Stefanie

Published
2022
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9. Author Correction: Osteosarcoma

Author

Beird, Hannah C., Bielack, Stefan S., Flanagan, Adrienne M., Gill, Jonathan, Heymann, Dominique, Janeway, Katherine A., Livingston, J. Andrew, Roberts, Ryan D., Strauss, Sandra J., and Gorlick, Richard

Published
2022
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10. Osteosarcoma

Author

Beird, Hannah C., Bielack, Stefan S., Flanagan, Adrianne M., Gill, Jonathan, Heymann, Dominique, Janeway, Katherine A., Livingston, J. Andrew, Roberts, Ryan D., Strauss, Sandra J., and Gorlick, Richard

Published
2022
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12. Supplementary Figures S1-S5 from Combined Immunotherapy Improves Outcome for Replication-Repair-Deficient (RRD) High-Grade Glioma Failing Anti–PD-1 Monotherapy: A Report from the International RRD Consortium

Author

Das, Anirban, primary, Fernandez, Nicholas R., primary, Levine, Adrian, primary, Bianchi, Vanessa, primary, Stengs, Lucie K., primary, Chung, Jiil, primary, Negm, Logine, primary, Dimayacyac, Jose Rafael, primary, Chang, Yuan, primary, Nobre, Liana, primary, Ercan, Ayse B., primary, Sanchez-Ramirez, Santiago, primary, Sudhaman, Sumedha, primary, Edwards, Melissa, primary, Larouche, Valerie, primary, Samuel, David, primary, Van Damme, An, primary, Gass, David, primary, Ziegler, David S., primary, Bielack, Stefan S., primary, Koschmann, Carl, primary, Zelcer, Shayna, primary, Yalon-Oren, Michal, primary, Campino, Gadi Abede, primary, Sarosiek, Tomasz, primary, Nichols, Kim E., primary, Loret De Mola, Rebecca, primary, Bielamowicz, Kevin, primary, Sabel, Magnus, primary, Frojd, Charlotta A., primary, Wood, Matthew D., primary, Glover, Jason M., primary, Lee, Yi-Yen, primary, Vanan, Magimairajan, primary, Adamski, Jenny K., primary, Perreault, Sebastien, primary, Chamdine, Omar, primary, Hjort, Magnus Aasved, primary, Zapotocky, Michal, primary, Carceller, Fernando, primary, Wright, Erin, primary, Fedorakova, Ivana, primary, Lossos, Alexander, primary, Tanaka, Ryuma, primary, Osborn, Michael, primary, Blumenthal, Deborah T., primary, Aronson, Melyssa, primary, Bartels, Ute, primary, Huang, Annie, primary, Ramaswamy, Vijay, primary, Malkin, David, primary, Shlien, Adam, primary, Villani, Anita, primary, Dirks, Peter B., primary, Pugh, Trevor J., primary, Getz, Gad, primary, Maruvka, Yosef E., primary, Tsang, Derek S., primary, Ertl-Wagner, Birgit, primary, Hawkins, Cynthia, primary, Bouffet, Eric, primary, Morgenstern, Daniel A., primary, and Tabori, Uri, primary

Published
2024
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13. Combined Immunotherapy Improves Outcome for Replication-Repair-Deficient (RRD) High-Grade Glioma Failing Anti–PD-1 Monotherapy: A Report from the International RRD Consortium

Author

Das, Anirban, primary, Fernandez, Nicholas R., additional, Levine, Adrian, additional, Bianchi, Vanessa, additional, Stengs, Lucie K., additional, Chung, Jiil, additional, Negm, Logine, additional, Dimayacyac, Jose Rafael, additional, Chang, Yuan, additional, Nobre, Liana, additional, Ercan, Ayse B., additional, Sanchez-Ramirez, Santiago, additional, Sudhaman, Sumedha, additional, Edwards, Melissa, additional, Larouche, Valerie, additional, Samuel, David, additional, Van Damme, An, additional, Gass, David, additional, Ziegler, David S., additional, Bielack, Stefan S., additional, Koschmann, Carl, additional, Zelcer, Shayna, additional, Yalon-Oren, Michal, additional, Campino, Gadi Abede, additional, Sarosiek, Tomasz, additional, Nichols, Kim E., additional, Loret De Mola, Rebecca, additional, Bielamowicz, Kevin, additional, Sabel, Magnus, additional, Frojd, Charlotta A., additional, Wood, Matthew D., additional, Glover, Jason M., additional, Lee, Yi-Yen, additional, Vanan, Magimairajan, additional, Adamski, Jenny K., additional, Perreault, Sebastien, additional, Chamdine, Omar, additional, Hjort, Magnus Aasved, additional, Zapotocky, Michal, additional, Carceller, Fernando, additional, Wright, Erin, additional, Fedorakova, Ivana, additional, Lossos, Alexander, additional, Tanaka, Ryuma, additional, Osborn, Michael, additional, Blumenthal, Deborah T., additional, Aronson, Melyssa, additional, Bartels, Ute, additional, Huang, Annie, additional, Ramaswamy, Vijay, additional, Malkin, David, additional, Shlien, Adam, additional, Villani, Anita, additional, Dirks, Peter B., additional, Pugh, Trevor J., additional, Getz, Gad, additional, Maruvka, Yosef E., additional, Tsang, Derek S., additional, Ertl-Wagner, Birgit, additional, Hawkins, Cynthia, additional, Bouffet, Eric, additional, Morgenstern, Daniel A., additional, and Tabori, Uri, additional

Published
2024
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14. Primary Multi-Systemic Metastases in Osteosarcoma: Presentation, Treatment, and Survival of 83 Patients of the Cooperative Osteosarcoma Study Group

Author

Mettmann, Vanessa L., primary, Blattmann, Claudia, additional, Friedel, Godehard, additional, Harrabi, Semi, additional, von Kalle, Thekla, additional, Kager, Leo, additional, Kevric, Matthias, additional, Kühne, Thomas, additional, Nathrath, Michaela, additional, Sorg, Benjamin, additional, Werner, Mathias, additional, Bielack, Stefan S., additional, and Hecker-Nolting, Stefanie, additional

Published
2024
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16. Osteosarcoma Arising After Ewing Sarcoma orVice Versa: A Report of 20 Affected Patients from the Cooperative Osteosarcoma Study Group (COSS)

Author

BIELACK, STEFAN S., primary, BLATTMANN, CLAUDIA, additional, HASSENPFLUG, WOLF, additional, KAGER, LEO, additional, KUHNE, THOMAS, additional, KEVRIC, MATTHIAS, additional, SCHLEGEL, PAUL-GERHARDT, additional, METTMANN, VANESSA, additional, SORG, BENJAMIN, additional, and HECKER-NOLTING, STEFANIE, additional

Published
2023
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17. Methotrexate, Doxorubicin, and Cisplatin (MAP) Plus Maintenance Pegylated Interferon Alfa-2b Versus MAP Alone in Patients With Resectable High-Grade Osteosarcoma and Good Histologic Response to Preoperative MAP: First Results of the EURAMOS-1 Good Response Randomized Controlled Trial

Author

Bielack, Stefan S, Smeland, Sigbjørn, Whelan, Jeremy S, Marina, Neyssa, Jovic, Gordana, Hook, Jane M, Krailo, Mark D, Gebhardt, Mark, Pápai, Zsuzsanna, Meyer, James, Nadel, Helen, Randall, R Lor, Deffenbaugh, Claudia, Nagarajan, Rajaram, Brennan, Bernadette, Letson, G Douglas, Teot, Lisa A, Goorin, Allen, Baumhoer, Daniel, Kager, Leo, Werner, Mathias, Lau, Ching C, Hall, Kirsten Sundby, Gelderblom, Hans, Meyers, Paul, Gorlick, Richard, Windhager, Reinhard, Helmke, Knut, Eriksson, Mikael, Hoogerbrugge, Peter M, Schomberg, Paula, Tunn, Per-Ulf, Kühne, Thomas, Jürgens, Heribert, van den Berg, Henk, Böhling, Tom, Picton, Susan, Renard, Marleen, Reichardt, Peter, Gerss, Joachim, Butterfass-Bahloul, Trude, Morris, Carol, Hogendoorn, Pancras CW, Seddon, Beatrice, Calaminus, Gabriele, Michelagnoli, Maria, Dhooge, Catharina, Sydes, Matthew R, and Bernstein, Mark

Subjects
Clinical Trials and Supportive Activities, Rare Diseases, Clinical Research, Cancer, Evaluation of treatments and therapeutic interventions, 6.1 Pharmaceuticals, Adolescent, Adult, Antineoplastic Combined Chemotherapy Protocols, Asia, Australia, Bone Neoplasms, Chemotherapy, Adjuvant, Child, Child, Preschool, Cisplatin, Disease Progression, Disease-Free Survival, Doxorubicin, Europe, Female, Humans, Interferon alpha-2, Interferon-alpha, Kaplan-Meier Estimate, Male, Methotrexate, Neoadjuvant Therapy, Neoplasm Grading, North America, Osteosarcoma, Osteotomy, Polyethylene Glycols, Proportional Hazards Models, Recombinant Proteins, Risk Factors, Time Factors, Treatment Outcome, Young Adult, EURAMOS-1 investigators, Clinical Sciences, Oncology and Carcinogenesis, Oncology & Carcinogenesis
Abstract

PurposeEURAMOS-1, an international randomized controlled trial, investigated maintenance therapy with pegylated interferon alfa-2b (IFN-α-2b) in patients whose osteosarcoma showed good histologic response (good response) to induction chemotherapy.Patients and methodsAt diagnosis, patients age ≤ 40 years with resectable high-grade osteosarcoma were registered. Eligibility after surgery for good response random assignment included ≥ two cycles of preoperative MAP (methotrexate, doxorubicin, and cisplatin), macroscopically complete surgery of primary tumor, < 10% viable tumor, and no disease progression. These patients were randomly assigned to four additional cycles MAP with or without IFN-α-2b (0.5 to 1.0 μg/kg per week subcutaneously, after chemotherapy until 2 years postregistration). Outcome measures were event-free survival (EFS; primary) and overall survival and toxicity (secondary).ResultsGood response was reported in 1,041 of 2,260 registered patients; 716 consented to random assignment (MAP, n = 359; MAP plus IFN-α-2b, n = 357), with baseline characteristics balanced by arm. A total of 271 of 357 started IFN-α-2b; 105 stopped early, and 38 continued to receive treatment at data freeze. Refusal and toxicity were the main reasons for never starting IFN-α-2b and for stopping prematurely, respectively. Median IFN-α-2b duration, if started, was 67 weeks. A total of 133 of 268 patients who started IFN-α-2b and provided toxicity information reported grade ≥ 3 toxicity during IFN-α-2b treatment. With median follow-up of 44 months, 3-year EFS for all 716 randomly assigned patients was 76% (95% CI, 72% to 79%); 174 EFS events were reported (MAP, n = 93; MAP plus IFN-α-2b, n = 81). Hazard ratio was 0.83 (95% CI, 0.61 to 1.12; P = .214) from an adjusted Cox model.ConclusionAt the preplanned analysis time, MAP plus IFN-α-2b was not statistically different from MAP alone. A considerable proportion of patients never started IFN-α-2b or stopped prematurely. Long-term follow-up for events and survival continues.

Published
2015

19. Beyond 5-year survival. A report from the Cooperative Osteosarcoma Study Group (COSS).

Author

Fernandes, Julia S., Blattmann, Claudia, Hecker-Nolting, Stefanie, Kager, Leo, Kevric, Matthias, Mettmann, Vanessa, Sorg, Benjamin, Fernandes, Marc, and Bielack, Stefan S.

Subjects
OSTEOSARCOMA, PROGNOSIS, SYMPTOMS, NEOADJUVANT chemotherapy, THERAPEUTICS
Abstract

Purpose: Prognostic factors have been well described for osteosarcoma, but analyses evaluating the further course of long-term survivors are lacking. We used the large database of the Cooperative Osteosarcoma Study Group (COSS) to perform such an analysis. Patients and Methods: The COSS database 1980-04/2019 was searched for 5-year survivors of primary high-grade central osteosarcoma of the extremities or trunk. Identified patients were analyzed for their further survival outcomes, assessing potentially prognostic and predictive factors already evident at initial disease presentation and treatment as well as their disease course during the first 5years of follow-up. Results: Two thousand and nine former eligible patients were identified (median age at initial diagnosis 15.1 (2.5–63.0) years; male vs. female 1149 (57.2%) vs. 860 (42.8%); extremities vs. trunk 1927 (95.9%) vs. 82 (4.1%); extremity primaries <1/3 vs. ≥1/3 of the involved bone 997 (67.8%) vs. 474 (32.2%) (456 unknown); localized vs. primary metastatic 1881 (93.6%) vs. 128 (6.4%); osteosarcoma as a secondary malignancy 41/2009 (2.0%)). Therapy starting by chemotherapy versus primary surgery 1860 (92.6%) versus 149 (7.4%); definitive tumor surgery by limb salvage versus ablative 1347 (67.0%) versus 659 (1 no surgery, 2 unknown); tumor response to preoperative chemotherapy documented for 1765 (94.9%) patients receiving neoadjuvant chemotherapy, good (<10% viable tumor) versus poor 1130 (64.0%) versus 635 (36.0%), local radiotherapy documented for 19 (0.9%) tumors. Recurrence during preceding 5years no versus yes 1681 (83.7%) versus 328 (16.3%). Median follow-up starting 5years after initial diagnosis 6.1 (0.002–32.2) years; 1815 survivors and 194 deaths. Overall survival after another 5/10/15/20years 91.7%/88.9%/85.8%/83.4% for all patients; 97.5%/95.2%/92.4%/89.9% if in remission years 1–5 versus 62.7%/57.3%/53.0%/51.2% if recurrence year 1–5 (p<0.001). Significant predictors of survival for all patients age at diagnosis (p=0.038), tumor site (p=0.030), having experienced the osteosarcoma as secondary malignancy (p<0.001), tumor response to preoperative chemotherapy (p=0.002). Multivariate Cox regression testing possible for 1759 (87.6%) patients with complete dataset: Having had a recurrence in years 1–5 (p<0.001), older age at diagnosis (p=0.009), and osteosarcoma as secondary malignancy (p=0.013) retained significance. Discussion: Highly important predictors of death such as the extent of tumor response to chemotherapy no longer remain valid after 5-year survival. The individual history of malignancies and their outcomes seems to gain pivotal importance. Conclusion: This benchmark analysis clearly defined risk factors for the further course of 5-year survivors from osteosarcoma. It argues for large disease-oriented databases as well as for very long follow-up periods. Novel findings will most likely require innovative statistical models to analyze such cohorts. [ABSTRACT FROM AUTHOR]

Published
2024
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20. Prognostic Factors in High-Grade Osteosarcoma of theExtremities or Trunk: An Analysis of 1,702 Patients Treatedon Neoadjuvant Cooperative Osteosarcoma Study GroupProtocols

Author

Bielack, Stefan S., primary, Kempf-Bielack, Beate, additional, Delling, Günter, additional, Exner, G. Ulrich, additional, Flege, Silke, additional, Helmke, Knut, additional, Kotz, Rainer, additional, Salzer-Kuntschik, Mechthild, additional, Werner, Matthias, additional, Winkelmann, Winfried, additional, Zoubek, Andreas, additional, Jürgens, Heribert, additional, and Winkler, Kurt, additional

Published
2023
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21. Solitary pulmonary metastases at first recurrence of osteosarcoma: Presentation, treatment, and survival of 219 patients of the Cooperative Osteosarcoma Study Group

Author

Mettmann, Vanessa L., primary, Baumhoer, Daniel, additional, Bielack, Stefan S., additional, Blattmann, Claudia, additional, Friedel, Godehard, additional, von Kalle, Thekla, additional, Kager, Leo, additional, Kevric, Matthias, additional, Nathrath, Michaela, additional, Sorg, Benjamin, additional, Dürken, Matthias, additional, and Hecker‐Nolting, Stefanie, additional

Published
2023
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22. Outcome of rare primary malignant bone sarcoma treated with multimodal therapy: Results from the EUROpean Bone Over 40 Sarcoma Study (EURO‐B.O.S.S.)

Author

Palmerini, Emanuela, primary, Reichardt, Peter, additional, Hall, Kirsten Sundby, additional, Bertulli, Rossella, additional, Bielack, Stefan S., additional, Comandone, Alessandro, additional, Egerer, Gerlinde, additional, Hansmeier, Anna, additional, Kevric, Matthias, additional, Carretta, Elisa, additional, Hansson, Lina, additional, Jebsen, Nina, additional, Eriksson, Mikael, additional, Bruland, Øyvind S., additional, Donati, Davide Maria, additional, Ibrahim, Toni, additional, Smeland, Sigbjørn, additional, and Ferrari, Stefano, additional

Published
2023
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23. Systemische Therapie von Osteosarkomen

Author

Bielack, Stefan S., Reichardt, Peter, Lindner, Lars H., and Hecker-Nolting, Stefanie

Abstract

Hintergrund: Osteosarkome sind die häufigsten primär malignen Knochentumorerkrankungen. Ihre gut etablierte Standardtherapie besteht aus einer kompletten Tumoroperation, die in eine intensive prä- und postoperative Chemotherapie eingebettet wird. Ziel: Es erfolgt die Beschreibung des Ist-Standards und aktueller Entwicklungen und Perspektiven der medikamentösen Tumortherapie in Abhängigkeit vom Patientenalter. Material und Methoden: Dabei handelt es sich um einen nichtsystematischen Literaturreview. Ergebnisse: Der Epidemiologie folgend, wurden fast alle Therapiestudien bei Kindern, Jugendlichen und jungen Erwachsenen durchgeführt. In den letzten Jahrzehnten ist in diesen allerdings eine Stagnation der Heilungsraten zu beobachten. Die Kombination der Chemotherapie mit einem Tyrosinkinasehemmer gilt als eine Möglichkeit, die diese Stagnation durchbrechen könnte. Noch stehen entsprechende Studien allerdings aus. Betroffene Erwachsene mittleren Alters profitieren ebenfalls von einer zusätzlich zur Lokaltherapie eingesetzten (neo)adjuvanten Chemotherapie. Die Auswahl der Medikamente und Dosierungen muss dabei an das Alter angepasst werden. Kaum etwas ist über noch ältere Patienten im Rentenalter bekannt, die konventionelle Chemotherapie i. d. R. schlecht tolerieren. Hier müssen, mangels aussagekräftiger Studien, individuelle Therapieangebote gefunden werden. Schlussfolgerung: Es ist zu hoffen, dass die raschen Erkenntnisgewinne zur Tumorbiologie auch bei Osteosarkompatienten jeden Alters zu relevanten therapeutischen Fortschritten führen werden.

Published
2024
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24. Comparison of MAPIE versus MAP in patients with a poor response to preoperative chemotherapy for newly diagnosed high-grade osteosarcoma (EURAMOS-1): an open-label, international, randomised controlled trial

Author

Marina, Neyssa M, Smeland, Sigbjørn, Bielack, Stefan S, Bernstein, Mark, Jovic, Gordana, Krailo, Mark D, Hook, Jane M, Arndt, Carola, van den Berg, Henk, Brennan, Bernadette, Brichard, Bénédicte, Brown, Ken L B, Butterfass-Bahloul, Trude, Calaminus, Gabriele, Daldrup-Link, Heike E, Eriksson, Mikael, Gebhardt, Mark C, Gelderblom, Hans, Gerss, Joachim, Goldsby, Robert, Goorin, Allen, Gorlick, Richard, Grier, Holcombe E, Hale, Juliet P, Hall, Kirsten Sundby, Hardes, Jendrik, Hawkins, Douglas S, Helmke, Knut, Hogendoorn, Pancras C W, Isakoff, Michael S, Janeway, Katherine A, Jürgens, Heribert, Kager, Leo, Kühne, Thomas, Lau, Ching C, Leavey, Patrick J, Lessnick, Stephen L, Mascarenhas, Leo, Meyers, Paul A, Mottl, Hubert, Nathrath, Michaela, Papai, Zsuzsanna, Randall, R Lor, Reichardt, Peter, Renard, Marleen, Safwat, Akmal Ahmed, Schwartz, Cindy L, Stevens, Michael C G, Strauss, Sandra J, Teot, Lisa, Werner, Mathias, Sydes, Matthew R, and Whelan, Jeremy S

Published
2016
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28. Primary osteosarcoma of the ribs: A report from the Cooperative Osteosarcoma Study Group

Author

Bodmer, Nicole, primary, Hecker‐Nolting, Stefanie, additional, Friedel, Godehard, additional, Blattmann, Claudia, additional, Kager, Leo, additional, Kessler, Torsten, additional, Kevric, Matthias, additional, Kühne, Thomas, additional, Mettmann, Vanessa, additional, Müller‐Abt, Peter, additional, Sorg, Benjamin, additional, Theobald, Matthias, additional, and Bielack, Stefan S., additional

Published
2023
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30. Ultra-Late Osteosarcoma Recurrences: An Analysis of 17 Cooperative Osteosarcoma Study Group Patients with a First Recurrence Detected More Than 10 Years After Primary Tumor Diagnosis

Author

Hecker-Nolting, Stefanie, primary, Kager, Leo, additional, Kühne, Thomas, additional, Baumhoer, Daniel, additional, Blattmann, Claudia, additional, Friedel, Godehard, additional, von Kalle, Thekla, additional, Kevric, Mathias, additional, Mayer-Steinacker, Regine, additional, Schwarz, Rudolf, additional, Sorg, Benjamin, additional, Wirth, Thomas, additional, and Bielack, Stefan S., additional

Published
2023
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31. The Clinical and Biological Landscape of Constitutional Mismatch Repair Deficiency: An IRRDC Study

Author

Ercan, Ayse Bahar, primary, Aronson, Melyssa, additional, Fernandez, Nicholas R., additional, Chang, Yuan, additional, Levine, Adrian, additional, Liu, Amy Zhihui, additional, Negm, Logine, additional, Edwards, Melissa, additional, Bianchi, Vanessa, additional, Stengs, Lucie, additional, Chung, Brian, additional, Al-Battashi, Abeer, additional, Reschke, Agnes, additional, Lion, Alex, additional, Ahmad, Alia, additional, Lassaletta, Alvaro, additional, Reddy, Alyssa, additional, Al-Darraji, Amir Fadhil, additional, Shah, Amish C., additional, Van Damme, An, additional, Bendel, Anne E., additional, Rashid, Aqeela, additional, Margol, Ashley S., additional, Kelly, Bethany L., additional, Pencheva, Bojana, additional, Heald, Brandie, additional, Anglin, Brianna Lemieux, additional, Crooks, Bruce, additional, Koschmann, Carl, additional, Gilpin, Catherine, additional, Porter, Christopher C., additional, Gass, David, additional, Samuel, David, additional, Ziegler, David S., additional, Blumenthal, Deborah T., additional, Kuo, Dennis John, additional, Hamideh, Dima, additional, Basel, Donald, additional, Khuong Quang, Dong Anh, additional, Stearns, Duncan, additional, Opocher, Enrico, additional, Carcellar, Fernando, additional, Feldman, Hagit Baris, additional, Toledano, Helen, additional, Winer, Ira S., additional, Scheers, Isabelle, additional, Fedorakova, Ivana, additional, Su, Jack M., additional, Barrios, Jaime Vengoechea, additional, Sterba, Jaroslav, additional, Knipstein, Jeffrey, additional, Hansford, Jordan R., additional, Gonzales-Santos, Julieta Rita, additional, Bhatia, Kanika, additional, Bielamowicz, Kevin J., additional, Minhas, Khurram, additional, Nichols, Kim E., additional, Cole, Kristina A., additional, Penney, Lynette, additional, Hjort, Magnus Aasved, additional, Sabel, Magnus, additional, Gil-da-Costa, Maria Joao, additional, Murray, Matthew J., additional, Miller, Matthew, additional, Blundell, Maude, additional, Massimino, Maura, additional, Al-Hussaini, Maysa, additional, Al-Jadiry, Mazin Faisal, additional, Comito, Melanie A., additional, Osborn, Michael, additional, Link, Michael P., additional, Zapotocky, Michal, additional, Ghalibafian, Mithra, additional, Shaheen, Najma, additional, Mushtaq, Naureen, additional, Waespe, Nicolas, additional, Hijiya, Nobuko, additional, Fuentes Bolanos, Noemi, additional, Ahmad, Olfat, additional, Chamdine, Omar, additional, Roy, Paromita, additional, Pichurin, Pavel N., additional, Nymen, Per, additional, Pearlman, Rachel, additional, Auer, Rebecca C., additional, Sukumaran, Reghu K., additional, Kebudi, Rejin, additional, Dvir, Rina, additional, Raphael, Robert, additional, Elhasid, Ronit, additional, McGee, Rose B., additional, Chami, Rose, additional, Noss, Ryan, additional, Tanaka, Ryuma, additional, Raskin, Salmo, additional, Sen, Santanu, additional, Lindhorst, Scott, additional, Perreault, Sebastian, additional, Caspi, Shani, additional, Riaz, Shazia, additional, Constantini, Shlomi, additional, Albert, Sophie, additional, Chaleff, Stanley, additional, Bielack, Stefan S., additional, Chiaravalli, Stefano, additional, Cramer, Stuart Louis, additional, Roy, Sumita, additional, Cahn, Suzanne, additional, Penna, Suzanne, additional, Hamid, Syed Ahmer, additional, Ghafoor, Tariq, additional, Imam, Uzma, additional, Larouche, Valerie, additional, MagimairajanIssai, Vanan, additional, Foulkes, William, additional, Lee, Yi Yen, additional, Maruvka, Yosef E., additional, Greer, Mary-Louise C., additional, Durno, Carol, additional, Shlien, Adam, additional, Ertl-Wagner, Birgit, additional, Villani, Anita, additional, Malkin, David, additional, Hawkins, Cynthia, additional, Bouffet, Eric, additional, Das, Anirban, additional, and Tabori, Uri, additional

Published
2023
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32. Next-generation personalised medicine for high-risk paediatric cancer patients – The INFORM pilot study

Author

Worst, Barbara C., van Tilburg, Cornelis M., Balasubramanian, Gnana Prakash, Fiesel, Petra, Witt, Ruth, Freitag, Angelika, Boudalil, Miream, Previti, Christopher, Wolf, Stephan, Schmidt, Sabine, Chotewutmontri, Sasithorn, Bewerunge-Hudler, Melanie, Schick, Matthias, Schlesner, Matthias, Hutter, Barbara, Taylor, Lenka, Borst, Tobias, Sutter, Christian, Bartram, Claus R., Milde, Till, Pfaff, Elke, Kulozik, Andreas E., von Stackelberg, Arend, Meisel, Roland, Borkhardt, Arndt, Reinhardt, Dirk, Klusmann, Jan-Henning, Fleischhack, Gudrun, Tippelt, Stephan, Dirksen, Uta, Jürgens, Heribert, Kramm, Christof M., von Bueren, Andre O., Westermann, Frank, Fischer, Matthias, Burkhardt, Birgit, Wößmann, Wilhelm, Nathrath, Michaela, Bielack, Stefan S., Frühwald, Michael C., Fulda, Simone, Klingebiel, Thomas, Koscielniak, Ewa, Schwab, Matthias, Tremmel, Roman, Driever, Pablo Hernáiz, Schulte, Johannes H., Brors, Benedikt, von Deimling, Andreas, Lichter, Peter, Eggert, Angelika, Capper, David, Pfister, Stefan M., Jones, David T.W., and Witt, Olaf

Published
2016
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33. Synovial Sarcoma Recurrence in Children and Young Adults

Author

Scheer, Monika, Dantonello, Tobias, Hallmen, Erika, Blank, Bernd, Sparber-Sauer, Monika, Vokuhl, Christian, Leuschner, Ivo, Münter, Marc W., von Kalle, Thekla, Bielack, Stefan S., Klingebiel, Thomas, Koscielniak, Ewa, and On behalf of the Cooperative Weichteilsarkom Studiengruppe [CWS]

Published
2016
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38. Secondary malignant neoplasms after bone and soft tissue sarcomas in children, adolescents, and young adults

Author

Kube, Stefanie J., primary, Blattmann, Claudia, additional, Bielack, Stefan S., additional, Kager, Leo, additional, Kaatsch, Peter, additional, Kühne, Thomas, additional, Sorg, Benjamin, additional, Kevric, Matthias, additional, Jabar, Susanne, additional, Hallmen, Erika, additional, Sparber‐Sauer, Monika, additional, Klingebiel, Thomas, additional, Koscielniak, Ewa, additional, Dirksen, Uta, additional, Hecker‐Nolting, Stefanie, additional, and Gerß, Joachim W. O., additional

Published
2022
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39. Osteosarcoma and Causes of Death. A Report of 1.520 Deceased Patients from the Cooperative Osteosarcoma Study Group (COSS)

Author

Bielack, Stefan S., primary, Blattmann, Claudia, additional, Borkhardt, Arndt, additional, Csóka, Monika, additional, Hassenpflug, Wolf, additional, Kabíčková, Edita, additional, Kager, Leo, additional, Kessler, Thorsten, additional, Kratz, Christian, additional, Kühne, Thomas, additional, Kevric, Matthias, additional, Lehrnbecher, Thomas, additional, Mayer-Steinacker, Regine, additional, Mettmann, Vanessa, additional, Metzler, Markus, additional, Reichardt, Peter, additional, Rossig, Claudia, additional, Sorg, Benjamin, additional, von Luettichau, Irene, additional, Windhager, Reinhard, additional, and Hecker-Nolting, Stefanie, additional

Published
2022
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41. Synovial sarcoma disease characteristics and primary tumor sites differ between patient age groups: a report of the Cooperative Weichteilsarkom Studiengruppe (CWS)

Author

Scheer, Monika, Blank, Bernd, Bauer, Sebastian, Vokuhl, Christian, Stegmaier, Sabine, Feuchtgruber, Simone, Henssen, Anton, Sparber-Sauer, Monika, Eggert, Angelika, Handgretinger, Ruppert, Pekrun, Arnulf, Rossig, Claudia, Rutkowski, Stefan, Schlegel, Paul-Gerhardt, Schrappe, Martin, Simon, Thorsten, Kazanowska, Bernarda, Niggli, Felix, Ladenstein, Ruth, Ljungman, Gustaf, Jahnukainen, Kirsi, Fuchs, Jörg, Bielack, Stefan S., Koscielniak, Ewa, Klingebiel, Thomas, and The Cooperative Weichteilsarkom Studiengruppe [CWS]

Subjects
Male, 0301 basic medicine, Cancer Research, medicine.medical_specialty, Adolescent, Medizin, Thigh, Gastroenterology, Sarcoma, Synovial, Young Adult, 03 medical and health sciences, 0302 clinical medicine, Patient age, Internal medicine, medicine, Humans, Neoplasm Invasiveness, Age of Onset, Neoplasm Metastasis, Child, Neoplasm Staging, Hematology, business.industry, Soft tissue sarcoma, Infant, General Medicine, medicine.disease, Primary tumor, Synovial sarcoma, 030104 developmental biology, medicine.anatomical_structure, Oncology, Disease Presentation, Child, Preschool, 030220 oncology & carcinogenesis, Female, Disease characteristics, business
Abstract

Older age is associated with worse outcome in synovial sarcoma (SS) patients. Differences in disease presentation among distinct age groups, however, are currently unknown.SS patients 21 years registered in consecutive CWS trials over the period of 1981-2018 were evaluated. Characteristics were analyzed according to age groups using the Fisher's exact test.The study population included 432 SS patients. Disease characteristics differed according to age groups of children (0-12 years, n = 176), adolescents (13-16 years, n = 178), and young adults (17-21 years, n = 78). The proportion of invasive tumors (T2) was significantly higher in older patients: children 33%, adolescents 39% and young adults 54%, p = 0.009805. Similarly, the proportion of tumors 10 cm was higher (13%, 21%, 31%; p = 0.005657) whereas conversely, the proportion of small tumors 3 cm was lower in older patients (29%, 24%, 6%; p = 0.000104). The presence of metastases at first diagnosis was also highest in older patients (6%, 10%, 21%, p = 0.000963). Notably, the proportion of thigh tumors was higher in older patients (p = 0.04173), whereas the proportion of head-neck tumors was lower in older patients (p = 0.08896).The rates of large, invasive tumors and the presence of metastases are significantly associated with older patient age. Localization to the thigh is more frequent in older patients.The causes for these variations require further exploration.

Published
2020

42. Current Insights into the Management of Late Chemotherapy Toxicities in Pediatric Osteosarcoma Patients

Author

Hecker-Nolting,Stefanie, Langer,Thorsten, Blattmann,Claudia, Kager,Leo, and Bielack,Stefan S

Subjects
Cancer Management and Research
Abstract

Stefanie Hecker-Nolting,1 Thorsten Langer,2 Claudia Blattmann,1 Leo Kager,3 Stefan S Bielack1,4 1Pädiatrie 5 (Onkologie, Hämatologie, Immunologie), Zentrum für Kinder-, Jugend- und Frauenmedizin, Stuttgart Cancer Center, Klinikum Stuttgart – Olgahospital, Stuttgart, Germany; 2Pädiatrische Onkologie und Hämatologie, Klinik für Kinder- und Jugendmedizin, Universitätsklinikum Schleswig-Holstein, Campus Lübeck, Lübeck, Germany; 3St. Anna Kinderspital, Abteilung für Kinder- und Jugendheilkunde, Medizinische Universität Wien, Vienna, Austria; 4Abteilung für Pädiatrische Hämatologie und Onkologie, Klinik für Kinder- und Jugendmedizin – Pädiatrische Hämatologie und Onkologie, Münster, GermanyCorrespondence: Stefan S BielackPädiatrie 5 (Onkologie, Hämatologie, Immunologie) Zentrum für Kinder-, Jugend- und Frauenmedizin, Stuttgart Cancer Center, Klinikum Stuttgart – Olgahospital, Kriegsbergstr. 62, Stuttgart, 70174, GermanyTel +49 711 2787 3881Fax +49 711 2787-3882Email coss@klinikum-stuttgart.deAbstract: With ever increasing long-term, disease free survival rates, long-term toxicities of otherwise successful therapy have gained increasing importance. They can be grouped into potentially life-threatening, especially secondary malignancies and anthracycline cardiomyopathies, potentially disabling, particularly severe hearing loss and renal insufficiency, other, and rare events. Pathophysiology, frequency, and medical treatment approaches are discussed. Finally, fertility issues and quality of life issues are discussed, together with an outlook into the future. The challenge to cure as many patients as possible from osteosarcoma while enabling a life free of late effects will remain.Keywords: osteosarcoma, child, adolescent, chemotherapy, late effects

Published
2021

43. Infantile myofibromatosis: Excellent prognosis but also rare fatal progressive disease. Treatment results of five Cooperative Weichteilsarkom Studiengruppe (CWS) trials and one registry

Author

Sparber‐Sauer, Monika, primary, Vokuhl, Christian, additional, Seitz, Guido, additional, Sorg, Benjamin, additional, Tobias, Möllers, additional, von Kalle, Thekla, additional, Münter, Marc, additional, Bielack, Stefan S., additional, Ladenstein, Ruth, additional, Ljungman, Gustaf, additional, Niggli, Felix, additional, Frühwald, Michael, additional, Loff, Stefan, additional, Klingebiel, Thomas, additional, and Koscielniak, Ewa, additional

Published
2021
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48. Tumour Volume Reduction After Neoadjuvant Chemotherapy Impacts Outcome in Localised Embryonal Rhabdomyosarcoma

Author

Dantonello, Tobias M., Stark, Monika, Timmermann, Beate, Fuchs, Jörg, Selle, Barbara, Linderkamp, Christin, Handgretinger, Rupert, Hagen, Rudolf, Feuchtgruber, Simone, Kube, Stefanie, Kosztyla, Daniel, Kazanowska, Bernarda, Ladenstein, Ruth, Niggli, Felix, Ljungman, Gustaf, Bielack, Stefan S., Klingebiel, Thomas, and Koscielniak, Ewa

Published
2015
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49. Long-term efficacy and safety of larotrectinib in an integrated dataset of patients with TRK fusion cancer.

Author

Hong, David S., primary, Shen, Lin, additional, van Tilburg, Cornelis Martinus, additional, Tan, Daniel Shao-Weng, additional, Kummar, Shivaani, additional, Lin, Jessica Jiyeong, additional, Doz, Francois P., additional, McDermott, Raymond S., additional, Albert, Catherine M., additional, Berlin, Jordan, additional, Bielack, Stefan S., additional, Lassen, Ulrik Niels, additional, Tahara, Makoto, additional, Norenberg, Ricarda, additional, Shurshalina, Anna, additional, Fellous, Marc Mardoche, additional, Nogai, Hendrik, additional, Xu, Rui-hua, additional, Laetsch, Theodore Willis, additional, and Drilon, Alexander E., additional

Published
2021
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