Primary metastatic Ewing's family tumors: results of the Italian Sarcoma Group and Scandinavian Sarcoma Group ISG/SSG IV Study including myeloablative chemotherapy and total-lung irradiation

Luksch, Roberto; Tienghi, A.; Hall, Kirsten Sundby; Fagioli, Franca; Picci, Piero; Barbieri, Enza; Gandola, Lorenza; Eriksson, Mikael; Ruggieri, Pietro; Daolio, Primo Andrea; Lindholm, Paula; Prete, Arcangelo; Bisogno, Gianni; Tamburini, Angela; Grignani, Giovanni; Abate, Massimo Eraldo; Podda, Marta; Smeland, Sigbjørn; Ferrari, Stefano

doi:10.1093/annonc/mds117

Cited by 88 publications

(106 citation statements)

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“…In landmark as well as in recent studies, improvement of local therapy has proven crucial in the treatment of advanced Ewing sarcoma patients [11, 20, 24], whereas the role of high-dose chemotherapy regimen with autologous stem cell transplantation in the treatment of advanced Ewing sarcoma patients remains subject to an ongoing discussion [4, 7, 10, 15–17, 25–35]. The conflicting results of various studies including our previous analyses [6, 20] in contrast to the results of Meyers et al [21] may be due to variation in intensity of local therapy.…”

Section: Discussionmentioning

confidence: 99%

Bone marrow involvement identifies a subgroup of advanced Ewing sarcoma patients with fatal outcome irrespective of therapy in contrast to curable patients with multiple bone metastases but unaffected marrow

et al. 2016

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PurposeAdvanced Ewing sarcomas have poor prognosis. They are defined by early relapse (<24 months after diagnosis) and/or by metastasis to multiple bones or bone marrow (BM). We analyzed risk factors, toxicity and survival in advanced Ewing sarcoma patients treated with the MetaEICESS vs. EICESS92 protocols.DesignOf 44 patients, 18 patients were enrolled into two subsequent MetaEICESS protocols between 1992 and 2014, and compared to outcomes of 26 advanced Ewing sarcoma patients treated with EICESS 1992 between 1992 and 1996. MetaEICESS 1992 consisted of induction chemotherapy, whole body imaging directed radiotherapy to the primary tumor and metastases, tandem high-dose chemotherapy and autologous rescue. In MetaEICESS 2007 this treatment was complemented by allogeneic stem cell transplantation. EICESS 1992 comprised induction chemotherapy, local therapy to the primary tumor only followed by consolidation chemotherapy.ResultsIn MetaEICESS 8/18 patients survived in complete remission vs. 2/26 in EICESS 1992 (p<0.05). Survival did not differ between MetaEICESS 2007 and MetaEICESS 1992. Three MetaEICESS patients died of complications, all in MetaEICESS 1992. After exclusion of patients succumbing to treatment related complications (n=3), 7/10 patients survived without BM involvement, in contrast to 0/5 patients with BM involvement. This was confirmed in a multivariate analysis. There was no correlation between BM involvement and the number of metastases at diagnosis.ConclusionThe MetaEICESS protocols yield long-term disease-free survival in patients with advanced Ewing sarcoma. Allogeneic stem cell transplantation was not associated with increased death of complications. Bone marrow involvement is a risk factor distinct from multiple bone metastases.

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Section: Discussionmentioning

confidence: 99%

Bone marrow involvement identifies a subgroup of advanced Ewing sarcoma patients with fatal outcome irrespective of therapy in contrast to curable patients with multiple bone metastases but unaffected marrow

et al. 2016

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“…The Italian Sarcoma Group and the Scandinavian Sarcoma Group designed a joint study to improve the prognosis for patients with Ewing's family tumors and synchronous metastatic disease limited to the lungs, or the pleura, or a single bone. 5-year event-free survival probability was 0.43 and the 5-year overall survival probability was 0.52 [12].…”

Section: Discussionmentioning

confidence: 93%

“…In patients with ES, an intensive approach with auto-HSCT is feasible and long-term survival is achievable in >50% of patients [9,12]. The Italian Sarcoma Group and the Scandinavian Sarcoma Group designed a joint study to improve the prognosis for patients with Ewing's family tumors and synchronous metastatic disease limited to the lungs, or the pleura, or a single bone.…”

Section: Discussionmentioning

confidence: 99%

Terapia wysokodawkowa z przeszczepieniem autologicznych komórek krwiotwórczych u dzieci z nerwiakiem zarodkowym i mięsakiem Ewinga

Styczyński

Dębski

Krenska

et al. 2017

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S u m m a r y B a c k g r o u n d . High-dose therapy (HDT) with autologous stem cell rescue has been recently applied in very-poor-risk pediatric solid tumors. Promising data have become available with the use of high-dose busulfan in neuroblastoma (NBL) and Ewing sarcoma (ES), and with high-dose treosulfan in ES. HDT approach resulted in an encouraging outcome without toxic mortality for high-risk patients.O b j e c t i v e . The objective of this study is to present transplant outcomes, that is disease-free-survival and overall survival in children with high-risk NBL and ES undergoing auto-HSCT. P a t i e n t s a n d m e t h o d s . A total number of 47 NBL and 20 ES auto-HSCT performed between 2004 and 2016 in a single transplant center were included in this analysis.R e s u l t s . Probability of 3-years pOS was 0.79±0.06 and 0.46±0.14 for NBL and ES patients, respectively. Relapse incidence at 3 years after HSCT was 0.37±0.08 and 0.26±0.11 for NBL and ES patients, respectively. The number of relapses at 3 years after HSCT was 15/47 in NBL and 6/20 in ES. Busulfan-based vs treosulfan-based conditioning in ES patients resulted in lower relapse and death rates. NBL and ES patients transplanted in complete remission (CR1) had lower relapse rates and lower death rates than patients at CR>1.C o n c l u s i o n. Obtained results of auto-HSCT confirm the therapeutic benefit for children with NBL and ES. Recent reports on current practice of HSCT in Europe indicate HDT with auto-HSCT as a standard of care in pediatric patients with high risk or relapsed NBL and ES. S t r e s z c z e n i e W s t ę p . Terapia wysokodawkowa (HDT) i transplantacja autologicznych komórek krwiotwórczych (auto-HSCT) została wprowadzona w leczeniu dzieci ze źle rokujacymi guzami litymi. Wstępne dane wskazują na wartość HDT opartej na busulfanie w neuroblastoma (NBL) i mięsaku Ewinga (ES) oraz zastosowaniu treosulfanu w ES. HDT z auto-HSCT stwarza możliwości poprawy wyników terapii przeciwnowotworowej, bez dodatkowej śmiertelności u pacjentów wysokiego ryzyka niepowodzenia terapeutycznego.C e l e m p r acy była analiza wyników terapii u dzieci z NBL i ES poddawanym HDT i auto-HSCT. W n i o s k i . Wyniki HDT z auto-HSCT potwierdzają korzyść terapeutyczną u dzieci z NBL i ES. Aktualne analizy europejskie wskazują, że takie postępowanie zostało uznane za standard opieki u dzieci z tymi chorobami.

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“…Beş yıllık genel sağkalım ve olaysız sağkalım oranları, sırası ile, %52 ve %43 olarak bildirilmiştir. [70] Nadir görülen malign kemik tümörleri Primer malign kemik tümörü tanısı olan 2627 hastayı içeren geniş bir analizde, osteosarkoma %35,1; kondrosarkom %25,8; Ewing sarkomu %16; kordoma %8,4 ve malign fibröz histiyositoma, fibrosarkom da dahil olmak üzere %5,6 olarak saptanmıştır. Beş yıllık genel sağkalım oranları ise osteosarkomda %41, kondrosarkomda %72,7; Ewing sarkomunda %41,2; kordomada %63,8 ve malign fibröz histositomda %42,9 olarak bildirilmiştir.…”

Section: Radyoterapiye Bağlı Yan Etkilerunclassified

Sık görülen primer malign kemik tümörlerinde radyoterapinin yeri ve tedaviye genel yaklaşım

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Primary metastatic Ewing's family tumors: results of the Italian Sarcoma Group and Scandinavian Sarcoma Group ISG/SSG IV Study including myeloablative chemotherapy and total-lung irradiation

Cited by 88 publications

References 31 publications

Bone marrow involvement identifies a subgroup of advanced Ewing sarcoma patients with fatal outcome irrespective of therapy in contrast to curable patients with multiple bone metastases but unaffected marrow

Bone marrow involvement identifies a subgroup of advanced Ewing sarcoma patients with fatal outcome irrespective of therapy in contrast to curable patients with multiple bone metastases but unaffected marrow

Terapia wysokodawkowa z przeszczepieniem autologicznych komórek krwiotwórczych u dzieci z nerwiakiem zarodkowym i mięsakiem Ewinga

Sık görülen primer malign kemik tümörlerinde radyoterapinin yeri ve tedaviye genel yaklaşım

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