Non-parameningeal head and neck rhabdomyosarcoma in children, adolescents, and young adults: Experience of the European paediatric Soft tissue sarcoma Study Group (EpSSG) - RMS2005 study.


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:
07 2021
Historique:
received: 17 02 2021
revised: 22 03 2021
accepted: 06 04 2021
pubmed: 11 5 2021
medline: 9 11 2021
entrez: 10 5 2021
Statut: ppublish

Résumé

The primary aim of this study was to analyse and evaluate the impact of different local treatments on the pattern of relapse in children with primary head and neck non-parameningeal (HNnPM) rhabdomyosarcoma (RMS), treated in the European paediatric Soft tissue sarcoma Study Group (EpSSG) RMS2005 study. The secondary aim was to assess whether current risk stratification is valid for this specific site. This study includes all patients with localised HNnPM RMS enrolled in the RMS2005 study between 2005 and 2016. Treatment comprised chemotherapy adapted to risk group, with local surgery and/or radiation therapy. The main outcome measures were event-free survival (EFS) and overall survival (OS). A total of 165 patients were identified; the median age was 6.4 years (range, 0.1-25). The most common tumour sites were cheek/chin (22%) and nasal ala/nasolabial fold (20%). Histology was unfavourable for 40%, and regional nodal involvement present in 26%. Local therapy included surgery (58%) and/or radiotherapy (72%) to primary tumour and/or regional lymph nodes. After a median follow-up of 66 months (range, 6-158), 42 patients experienced an event, and 17 are still alive. Tumour events were frequent in oral primary (36%), parotid site (26%), cheek/chin (24%), and nasal ala/nasolabial fold (24%) and included locoregional failure in 84% of cases. The 5-year EFS and OS were 75% (95% confidence interval [CI]: 67.3-81.2) and 84.9% (95% CI: 77.5-89.7), respectively. Favourable histology was associated with a better EFS (82.3% versus 64.6%; p = 0.02) and nodal spread with a worse OS (88.6% versus 76.1%; p = 0.04). Different sublocations within the HNnPM primary did not have significant impact on outcome. Locoregional relapse/progression is the main tumour failure event in this site. Despite frequent unfavourable risk factors, HNnPM RMS remains a favourable location in the context of a risk-adapted strategy.

Sections du résumé

BACKGROUND/OBJECTIVES
The primary aim of this study was to analyse and evaluate the impact of different local treatments on the pattern of relapse in children with primary head and neck non-parameningeal (HNnPM) rhabdomyosarcoma (RMS), treated in the European paediatric Soft tissue sarcoma Study Group (EpSSG) RMS2005 study. The secondary aim was to assess whether current risk stratification is valid for this specific site.
DESIGN/METHODS
This study includes all patients with localised HNnPM RMS enrolled in the RMS2005 study between 2005 and 2016. Treatment comprised chemotherapy adapted to risk group, with local surgery and/or radiation therapy. The main outcome measures were event-free survival (EFS) and overall survival (OS).
RESULTS
A total of 165 patients were identified; the median age was 6.4 years (range, 0.1-25). The most common tumour sites were cheek/chin (22%) and nasal ala/nasolabial fold (20%). Histology was unfavourable for 40%, and regional nodal involvement present in 26%. Local therapy included surgery (58%) and/or radiotherapy (72%) to primary tumour and/or regional lymph nodes. After a median follow-up of 66 months (range, 6-158), 42 patients experienced an event, and 17 are still alive. Tumour events were frequent in oral primary (36%), parotid site (26%), cheek/chin (24%), and nasal ala/nasolabial fold (24%) and included locoregional failure in 84% of cases. The 5-year EFS and OS were 75% (95% confidence interval [CI]: 67.3-81.2) and 84.9% (95% CI: 77.5-89.7), respectively. Favourable histology was associated with a better EFS (82.3% versus 64.6%; p = 0.02) and nodal spread with a worse OS (88.6% versus 76.1%; p = 0.04). Different sublocations within the HNnPM primary did not have significant impact on outcome.
CONCLUSION
Locoregional relapse/progression is the main tumour failure event in this site. Despite frequent unfavourable risk factors, HNnPM RMS remains a favourable location in the context of a risk-adapted strategy.

Identifiants

pubmed: 33971448
pii: S0959-8049(21)00228-8
doi: 10.1016/j.ejca.2021.04.007
pii:
doi:

Banques de données

EudraCT
['2005-000217-35']

Types de publication

Clinical Trial Comparative Study Journal Article Multicenter Study Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

84-93

Subventions

Organisme : Cancer Research UK
Pays : United Kingdom

Informations de copyright

Copyright © 2021 The Author(s). Published by Elsevier Ltd.. All rights reserved.

Déclaration de conflit d'intérêts

Conflict of interest statement The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Auteurs

Heidi Glosli (H)

Paediatric Research Institute, Division of Paediatric and Adolescent Medicine, Oslo University Hospital, Oslo, Norway. Electronic address: hglosli@ous-hf.no.

Gianni Bisogno (G)

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

Anna Kelsey (A)

Department of Paediatric Histopathology, Royal Manchester Children's Hospital, Manchester, United Kingdom.

Julia C Chisholm (JC)

Children and Young Peoples Unit, Royal Marsden Hospital, Down's Road, Sutton, Surrey SM2 5PT, United Kingdom.

Mark Gaze (M)

Department of Oncology, University College London Hospitals NHS Foundation Trust, London, United Kingdom.

Frederic Kolb (F)

Department of Plastic Surgery, Gustave Roussy Cancer Campus, Villejuif, France; Department of Plastic Surgery, UCSD, San Diego, CA, USA.

Kieran McHugh (K)

Department of Radiology, Great Ormond Street Hospital for Children, London, United Kingdom.

Janet Shipley (J)

The Institute of Cancer Research, Sutton, United Kingdom.

Soledad Gallego (S)

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

Johannes H M Merks (JHM)

Princess Máxima Center for Paediatric Oncology, Utrecht, the Netherlands.

Ludi E Smeele (LE)

Department of Head and Neck Oncology and Surgery, Netherlands Cancer Institute/Antoni van Leeuwenhoek Hospital, Amsterdam, the Netherlands; Department of Oral and Maxillofacial Surgery, Amsterdam UMC, Amsterdam, the Netherlands; Solid Tumor Department, Princess Máxima Center for Paediatric Oncology, Utrecht, the Netherlands.

Henry Mandeville (H)

Children and Young Peoples Unit & Haemato-oncology Unit, Royal Marsden Hospital, NHS Foundation Trust, Sutton, United Kingdom.

Andrea Ferrari (A)

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

Veronique Minard-Colin (V)

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

Nadege Corradini (N)

Paediatric Hematology and Oncology Institute, Léon Bérard Center, Lyon, France.

Meriel Jenney (M)

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

Ilaria Zanetti (I)

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

Gian L De Salvo (GL)

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

Daniel Orbach (D)

SIREDO Oncology Center, Institute Curie, PSL University, Paris, France.

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