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Pediatric Patients with Stage IV Rhabdomyosarcoma Significantly Benefit from Long-Term Maintenance Therapy: Results of the CWS-IV 2002 and the CWS DOK IV 2004-Trials
被引:13
作者:
Tramsen, Lars
[1
,2
]
Bochennek, Konrad
[2
]
Sparber-Sauer, Monika
[3
,4
]
Salzmann-Manrique, Emilia
[2
]
Scheer, Monika
[5
]
Dantonello, Tobias
[6
]
Borkhardt, Arndt
[7
]
Dirksen, Uta
[8
,9
]
Thorwarth, Anne
[5
]
Greiner, Jeanette
[10
]
Ebinger, Martin
[11
]
Weclawek-Tompol, Jadwiga
[12
]
Ladenstein, Ruth
[13
]
Ljungman, Gustaf
[14
]
Hallmen, Erika
[3
]
Lehrnbecher, Thomas
[2
]
Koscielniak, Ewa
[3
,4
]
Klingebiel, Thomas
[2
]
机构:
[1] Univ Hosp Schleswig Holstein, Dept Pediat, Campus Kiel, D-24105 Kiel, Germany
[2] Goethe Univ, Univ Hosp, Dept Children & Adolescents, D-60590 Frankfurt, Germany
[3] Olgahosp Stuttgart Canc Ctr, Hosp State Capital Stuttgart, Ctr Pediat Adolescent & Womens Med, Pediat Oncol Hematol Immunology 5, D-70174 Stuttgart, Germany
[4] Univ Tubingen, Fac Med, D-72016 Tubingen, Germany
[5] Charite Univ Med Berlin, Dept Pediat Oncol & Hematol, Augustenburger Pl 1, D-13353 Berlin, Germany
[6] Univ Bern, Bern Univ Hosp, Dept Pediat, Div Pediat Hematol & Oncol, CH-3010 Bern, Switzerland
[7] Heinrich Heine Univ, Med Fac, Dept Pediat Oncol Hematol & Clin Immunol, D-40225 Dusseldorf, Germany
[8] Univ Hosp Essen, West German Canc Ctr, Pediat 3, D-45147 Essen, Germany
[9] Natl Ctr Tumor Dis NCT Site Essen, German Canc Consortium Site Essen, D-45147 Essen, Germany
[10] Kantonsspital Aarau AG, Childrens Hosp, Pediat Oncol & Hematol, CH-5001 Aarau, Switzerland
[11] Univ Childrens Hosp, Dept Gen Pediat & Pediat Oncol & Hematol, D-72076 Tubingen, Germany
[12] Univ Med Wroclaw, Dept Bone Marrow Transplantat, Pediat Oncol & Haematol, Wroclaw, Poland
[13] St Anna Childrens Hosp, Childrens Canc Res Inst CCRI, A-1090 Vienna, Austria
[14] Uppsala Univ, Dept Womens & Childrens Hlth, Pediat Oncol, S-75185 Uppsala, Sweden
来源:
关键词:
soft tissue sarcoma;
rhabdomyosarcoma;
metastatic;
children;
long-term maintenance therapy;
outcome;
SOFT-TISSUE SARCOMA;
HIGH-DOSE CHEMOTHERAPY;
STEM-CELL TRANSPLANTATION;
PHASE-II WINDOW;
METASTATIC RHABDOMYOSARCOMA;
PROGNOSTIC-FACTORS;
POOLED ANALYSIS;
CHILDHOOD;
CHILDREN;
ONCOLOGY;
D O I:
10.3390/cancers15072050
中图分类号:
R73 [肿瘤学];
学科分类号:
100214 ;
摘要:
Rhabdomyosarcoma (RMS) is the most common soft tissue sarcoma (STS) in childhood. Whereas more than 90% of patients with localized low-risk RMS can be cured, metastatic RMS have a dismal outcome, with survival rates of less than 30%. The HD CWS-96 trial showed an improved outcome for patients receiving maintenance therapy after completing intensive chemotherapy. Consequently, the international clinical trials CWS-IV 2002 and CWS DOK IV 2004 on metastatic disease of STS of the Cooperative Weichteilsarkom Studiengruppe (CWS) were designed in addition to the CWS-2002P trial for localized RMS disease. All patients received a multimodal intensive treatment regimen. To maintain remission, three options were compared: long-term maintenance therapy (LTMT) versus allogeneic hematopoietic stem cell transplantation (alloHSCT) versus high-dose chemotherapy (HDCT). A total of 176 pediatric patients with a histologically confirmed diagnosis of metastatic RMS or RMS-like tumor were included. A total of 89 patients receiving LTML showed a significantly better outcome, with an event-free survival (EFS) of 41% and an overall survival (OS) of 53%, than alloHSCT (n = 21, EFS 19%, p = 0.02, OS 24%, p = 0.002). The outcome of LTML was slightly improved compared to HDCT (n = 13, EFS 35%, OS 34%). In conclusion, our data suggest that in patients suffering from metastatic RMS, long-term maintenance therapy is a superior strategy in terms of EFS and OS compared to alloHSCT. EFS and OS of HDCT are similar in these strategies; however, the therapeutic burden of LTMT is much lower.
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页数:14
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