Alveolar rhabdomyosarcoma with regional nodal involvement: Results of a combined analysis from two cooperative groups

被引：19

作者：

Gallego, Soledad ^{[1
]}

Chi, Yueh-Yun ^{[2
]}

De Salvo, Gian Luca ^{[3
]}

Li, Minjie ^{[4
]}

Merks, Johannes Hm ^{[5
]}

Rodeberg, David A. ^{[6
]}

Scheltinga, Sheila Terwisscha ^{[5
]}

Mascarenhas, Leo ^{[2
]}

Orbach, Daniel ^{[7
]}

Jenney, Meriel ^{[8
]}

Million, Lynn ^{[9
]}

Minard-Colin, Veronique ^{[10
]}

Wolden, Suzanne ^{[11
]}

Zanetti, Ilaria ^{[12
]}

Parham, David M. ^{[2
]}

Mandeville, Henry ^{[13
]}

Venkatramani, Rajkumar ^{[14
]}

Bisogno, Gianni ^{[12
]}

Hawkins, Douglas S. ^{[15
]}

机构：

[1] Univ Hosp Vall dHebron, P Vall dHebron 119-129, Barcelona 08035, Spain

[2] Univ Southern Calif, Childrens Hosp Los Angeles, Los Angeles, CA 90007 USA

[3] Veneto Oncol Inst IOV IRCCS, Clin Trials & Biostat Unit, Padua, Italy

[4] Univ Florida, Gainesville, FL USA

[5] Princess Maxima Ctr Pediat Oncol, Utrecht, Netherlands

[6] East Carolina Univ, Greenville, NC 27858 USA

[7] PSL Univ, Inst Curie, SIREDO Oncol Ctr, Paris, France

[8] Cardiff & Vale Univ Hlth Board, Children Hosp Wales, Cardiff, Wales

[9] Stanford Univ, Sch Med, Stanford, CA USA

[10] Gustave Roussy Univ Paris Saclay, Villejuif, France

[11] Mem Sloan Kettering Canc Ctr, 1275 York Ave, New York, NY 10021 USA

[12] Padova Univ Hosp, Padua, Italy

[13] Royal Marsden NHS Fdn Trust, Sutton, Surrey, England

[14] Texas Childrens Hosp, Baylor Coll Med, Houston, TX 77030 USA

[15] Univ Washington, Fred Hutchinson Canc Res Ctr, Seattle Childrens Hosp, Seattle, WA 98195 USA

来源：

PEDIATRIC BLOOD & CANCER | 2021年 / 68卷 / 03期

关键词：

alveolar rhabdomyosarcoma; chemotherapy; nodal involvement; prognostic factors; rhabdomyosarcoma;

D O I：

10.1002/pbc.28832

中图分类号：

R73 [肿瘤学];

学科分类号：

100214 ;

摘要：

Background: Treatment of children and adolescents with alveolar rhabdomyosarcoma (ARMS) and regional nodal involvement (N1) have been approached differently by North American and European cooperative groups. In order to define a better therapeutic strategy, we analyzed two studies conducted between 2005 and 2016 by the European paediatric Soft tissue sarcoma Study Group (EpSSG) and Children's Oncology Group (COG). Methods: We retrospectively identified patients with ARMS N1 enrolled in either EpSSG RMS2005 or in COG ARST0531. Chemotherapy in RMS2005 comprised ifosfamide + vincristine + dactinomycin + doxorubicin (IVADo), IVA and maintenance (vinorelbine, cyclophosphamide); in ARST0531, it consisted of either vincristine + dactinomycin + cyclophosphamide (VAC) or VAC alternating with vincristine + irinotecan (VI). Local treatment was similar in both protocols. Results: The analysis of the clinical characteristics of 239 patients showed some differences between study groups: in RMS2005, advanced Intergroup Rhabdomyosarcoma Study Group (IRS) and large tumors predominated. There were no differences in outcomes between the two groups: 5-year event-free survival (EFS), 49% (95% confidence interval [CI]: 39-59) and 44% (95% CI: 30-58), and overall survival (OS), 51% (95% CI: 41-61) and 53.6% (95% CI: 40-68) in RMS2005 and ARST0531, respectively. In RMS2005, EFS of patients with FOXO1-positive tumors was significantly inferior to those with FOXO1-negative (49.3% vs 73%, P = .034). In contrast, in ARST0531, EFS of patients with FOXO1-positive tumors was 45% compared with 43.8% for those with FOXO1-negative. Conclusions: The outcome of patients with ARMS N1 was similar in both protocols. However, patients with FOXO1 fusion-negative tumors enrolled in RMS2005 showed a significantly better outcome, suggesting that different strategies of chemotherapy may have an impact in the outcome of this subgroup of patients.

引用

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