Embryonal rhabdomyosarcoma completely resected at diagnosis: The European paediatric Soft tissue sarcoma Study Group RMS2005 experience.


Journal

European journal of cancer (Oxford, England : 1990)
ISSN: 1879-0852
Titre abrégé: Eur J Cancer
Pays: England
ID NLM: 9005373

Informations de publication

Date de publication:
03 2021
Historique:
received: 19 09 2020
revised: 12 11 2020
accepted: 18 12 2020
pubmed: 11 2 2021
medline: 29 9 2021
entrez: 10 2 2021
Statut: ppublish

Résumé

Rhabdomyosarcoma (RMS) is the most common form of soft tissue sarcoma in children. We report the results of the European paediatric Soft tissue sarcoma Study Group (EpSSG) RMS 2005 study, which prospectively evaluated the reduction of chemotherapy in patients with embryonal RMS (ERMS) after initial surgery. Between October 2005 and December 2016, all patients with localised ERMS with an initial microscopically complete resection (IRS group I) with lymph node-negative (N0) were prospectively enrolled in the low-risk (n = 70, subgroup A; age < 10 years and tumour size ≤ 5 cm) or standard-risk group (n = 108, subgroup B; age ≥ 10 years or tumour size > 5 cm. Subgroup A received 8 courses of vincristine and dactinomycin (VA) for 22 weeks; subgroup B received 4 courses of VA with ifosfamide (IVA) and 5 courses of VA for 25 weeks. The 5-year event-free survival (EFS) and overall survival (OS) were 90.8% (95% confidence interval [CI]: 85.0-94.4) and 95.7% (95% CI: 90.5-98.1), respectively (n = 178). The EFS and OS were 95.5% (95% CI: 86.8-98.5) and 100% (subgroupA), and 87.8% (95% CI: 79.3-93.0) and 93.0% (95% CI: 84.8-96.8)(subgroup B), respectively. Bearman stage 2 veno-occlusive disease (VOD) occurred in 4 very young patients. VA treatment for 8 courses was effective and well tolerated by the subgroup of patients with low-risk ERMS (group A). Four courses of IVA and 5 courses of VA instead of 9 courses of IVA also has very good results. Careful monitoring for liver toxicity is important in very young patients. European union drug regulating authorities clinical trials EUDRACT No. 2005-000217-35.

Sections du résumé

BACKGROUND
Rhabdomyosarcoma (RMS) is the most common form of soft tissue sarcoma in children. We report the results of the European paediatric Soft tissue sarcoma Study Group (EpSSG) RMS 2005 study, which prospectively evaluated the reduction of chemotherapy in patients with embryonal RMS (ERMS) after initial surgery.
METHODS
Between October 2005 and December 2016, all patients with localised ERMS with an initial microscopically complete resection (IRS group I) with lymph node-negative (N0) were prospectively enrolled in the low-risk (n = 70, subgroup A; age < 10 years and tumour size ≤ 5 cm) or standard-risk group (n = 108, subgroup B; age ≥ 10 years or tumour size > 5 cm. Subgroup A received 8 courses of vincristine and dactinomycin (VA) for 22 weeks; subgroup B received 4 courses of VA with ifosfamide (IVA) and 5 courses of VA for 25 weeks.
RESULTS
The 5-year event-free survival (EFS) and overall survival (OS) were 90.8% (95% confidence interval [CI]: 85.0-94.4) and 95.7% (95% CI: 90.5-98.1), respectively (n = 178). The EFS and OS were 95.5% (95% CI: 86.8-98.5) and 100% (subgroupA), and 87.8% (95% CI: 79.3-93.0) and 93.0% (95% CI: 84.8-96.8)(subgroup B), respectively. Bearman stage 2 veno-occlusive disease (VOD) occurred in 4 very young patients.
CONCLUSION
VA treatment for 8 courses was effective and well tolerated by the subgroup of patients with low-risk ERMS (group A). Four courses of IVA and 5 courses of VA instead of 9 courses of IVA also has very good results. Careful monitoring for liver toxicity is important in very young patients. European union drug regulating authorities clinical trials EUDRACT No. 2005-000217-35.

Identifiants

pubmed: 33567392
pii: S0959-8049(20)31450-7
doi: 10.1016/j.ejca.2020.12.025
pii:
doi:

Substances chimiques

Dactinomycin 1CC1JFE158
Vincristine 5J49Q6B70F
Doxorubicin 80168379AG
Cyclophosphamide 8N3DW7272P
Ifosfamide UM20QQM95Y

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

21-29

Investigateurs

Adriana Rose (A)
Christine Devalck (C)
Sima Ferman (S)
Peter Mudry (P)
Christophe Bergeron (C)
Myriam Weyl Ben Arush (MWB)
Gianni Bisogno (G)
Heidi Glosli (H)
Daniela Sejnova (D)
Maja Cesen (M)
Soledad Gallego (S)
J Hans Merks (JH)
Meriel Jenney (M)

Informations de copyright

Copyright © 2021 Elsevier Ltd. All rights reserved.

Déclaration de conflit d'intérêts

Conflict of interest statement The authors do not have competing interests.

Auteurs

Christophe Bergeron (C)

Department of Paediatric Oncology, Centre Léon Bérard, Lyon, France. Electronic address: christophe.bergeron@lyon.unicancer.fr.

Meriel Jenney (M)

Department of Paediatric Oncology, Children's Hospital for Wales, Cardiff, United Kingdom.

Federica De Corti (F)

Pediatric Surgery, Department of Women's and Children's Health, University-Hospital of Padova, Padova, Italy.

Soledad Gallego (S)

Paediatric Oncology, Hospital Universitari Vall D'Hebron, Barcelona, Spain.

Hans Merks (H)

Department of Paediatric Oncology, Emma Children's Hospital/Academic Medical Centre, Amsterdam, the Netherlands.

Heidi Glosli (H)

Department of Paediatric and Adolescent Medicine, Oslo University Hospital, Oslo, Norway.

Andrea Ferrari (A)

Pediatric Oncology Unit, Fondazione IRCCS Istituto Nazionale Tumori, Milan, Italy.

Dominique Ranchère-Vince (D)

Pathology Department, Centre Léon Bérard, Lyon, France.

Gian Luca De Salvo (GL)

Clinical Trials and Biostatistics Unit, Istituto Oncologico Veneto - IRCCS, Padova, Italy.

Ilaria Zanetti (I)

Hematology Oncology Division, Department of Women's and Children's Health, University of Padova, Padova, Italy.

Julia Chisholm (J)

Children and Young Peoples Unit, Royal Marsden Hospital, Surrey, United Kingdom.

Véronique Minard-Colin (V)

Department of Pediatric and Adolescent Oncology, Gustave-Roussy, Campus, Université Paris Saclay, Villejuif, France.

Timothy Rogers (T)

Department of Paediatric Surgery, University Hospitals Bristol NHS Foundation Trust, Bristol, United Kingdom.

Gianni Bisogno (G)

Hematology Oncology Division, Department of Women's and Children's Health, University of Padova, Padova, Italy.

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Classifications MeSH