Alveolar rhabdomyosarcoma with regional nodal involvement: Results of a combined analysis from two cooperative groups.
alveolar rhabdomyosarcoma
chemotherapy
nodal involvement
prognostic factors
rhabdomyosarcoma
Journal
Pediatric blood & cancer
ISSN: 1545-5017
Titre abrégé: Pediatr Blood Cancer
Pays: United States
ID NLM: 101186624
Informations de publication
Date de publication:
03 2021
03 2021
Historique:
received:
28
09
2020
revised:
26
10
2020
accepted:
08
11
2020
pubmed:
28
11
2020
medline:
3
8
2021
entrez:
27
11
2020
Statut:
ppublish
Résumé
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). 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. 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. 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.
Sections du résumé
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.
Identifiants
pubmed: 33245207
doi: 10.1002/pbc.28832
pmc: PMC8414760
mid: NIHMS1734154
doi:
Types de publication
Journal Article
Multicenter Study
Research Support, N.I.H., Extramural
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
e28832Subventions
Organisme : NIH HHS
ID : U10CA098543
Pays : United States
Organisme : Cancer Research UK
ID : 5943
Pays : United Kingdom
Organisme : NCI NIH HHS
ID : P30 CA008748
Pays : United States
Organisme : NIH HHS
ID : U10CA098413
Pays : United States
Organisme : NCI NIH HHS
ID : U10 CA098543
Pays : United States
Organisme : NCI NIH HHS
ID : U10 CA180899
Pays : United States
Organisme : NCI NIH HHS
ID : U10 CA180886
Pays : United States
Organisme : NCI NIH HHS
ID : U10 CA098413
Pays : United States
Organisme : NIH HHS
ID : U10CA180899
Pays : United States
Organisme : NIH HHS
ID : U10CA180886
Pays : United States
Informations de copyright
© 2020 Wiley Periodicals LLC.
Références
Eur J Cancer. 1998 Jun;34(7):1050-62
pubmed: 9849454
Pediatr Blood Cancer. 2014 Jan;61(1):53-67
pubmed: 23940101
J Clin Oncol. 2012 May 10;30(14):1670-7
pubmed: 22454413
J Clin Oncol. 2006 Aug 20;24(24):3844-51
pubmed: 16921036
J Clin Oncol. 2011 Apr 1;29(10):1319-25
pubmed: 21357778
J Clin Oncol. 2012 Jul 10;30(20):2457-65
pubmed: 22665534
J Clin Oncol. 2002 Jun 1;20(11):2672-9
pubmed: 12039929
Pediatr Blood Cancer. 2016 Apr;63(4):634-9
pubmed: 26756883
J Clin Oncol. 2011 Apr 1;29(10):1304-11
pubmed: 21357792
J Clin Oncol. 2013 Sep 10;31(26):3226-32
pubmed: 23940218
Cancer Med. 2019 Oct;8(14):6437-6448
pubmed: 31456361
J Clin Oncol. 2008 May 10;26(14):2384-9
pubmed: 18467730
Pediatr Blood Cancer. 2013 Sep;60(9):1411-7
pubmed: 23526739
J Pediatr Hematol Oncol. 2001 May;23(4):215-20
pubmed: 11846299
Cancer. 2018 Aug 1;124(15):3201-3209
pubmed: 29797665
Pediatr Blood Cancer. 2004 Jan;42(1):64-73
pubmed: 14752797
Eur J Surg Oncol. 2018 Jan;44(1):170-177
pubmed: 29208319
J Clin Oncol. 2016 Jan 10;34(2):117-22
pubmed: 26503200
Lancet Oncol. 2019 Nov;20(11):1566-1575
pubmed: 31562043
Cancer. 1995 Sep 15;76(6):1073-85
pubmed: 8625211
Pediatr Blood Cancer. 2017 Dec;64(12):
pubmed: 28521080
J Clin Oncol. 2009 Jul 10;27(20):3391-7
pubmed: 19398574
J Clin Oncol. 2018 Sep 20;36(27):2770-2777
pubmed: 30091945
Lancet Oncol. 2018 Aug;19(8):1061-1071
pubmed: 29941280
Eur J Cancer. 2015 Jan;51(2):193-201
pubmed: 25471261
J Clin Oncol. 2004 Dec 1;22(23):4787-94
pubmed: 15570080
J Pediatr Surg. 2000 Feb;35(2):317-21
pubmed: 10693687
Nat Rev Dis Primers. 2019 Jan 7;5(1):1
pubmed: 30617281
Cancer. 2019 Sep 15;125(18):3242-3248
pubmed: 31174239