Localised rhabdomyosarcoma in infants (<12 months) and young children (12-36 months of age) treated on the EpSSG RMS 2005 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:
01 2022
Historique:
received: 20 08 2021
revised: 11 10 2021
accepted: 24 10 2021
pubmed: 7 12 2021
medline: 29 1 2022
entrez: 6 12 2021
Statut: ppublish

Résumé

Infants (<12 months) with rhabdomyosarcoma have historically had poorer outcome than the older age groups. We present outcomes for infants and young children aged 12-36 months with localised rhabdomyosarcoma with a particular emphasis on infants. All children less than 36 months of age enrolled on the EpSSG RMS 2005 study for localised disease are included. Treatment comprised chemotherapy, local surgery and/or radiation therapy adapted to risk group and age. Main outcome measures were event free survival (EFS) and overall survival (OS). Outcome data were available for 485/490 patients aged less than 36 months, 110 were infants. Infants received chemotherapy according to the risk group with no toxic deaths. Radiotherapy was delivered to 33.6% of infants and 63.5% of 12-36 months old, with respectively 41.7% and 22.2% receiving brachytherapy. Radical surgery was performed in 62% of infants and 57.1% of 12-36 months old. Median follow up for patients who are alive (n = 393) was 72.7 months (range 6.9-158.2). Five-year OS for infants was 88.4% (95%CI 80.3-93.2), which is significantly better than the OS in 12-36 months old patients of 78.0% (95%CI 73.2-82.0; p = 0.0204). Five-year EFS for infants was 72.5% (95%CI 62.8-80.0) compared with 66.1% (95%CI 61.0-70.7; p = 0.2663) for 12-36 months old. Infants treated on RMS 2005 achieved excellent EFS and OS. The EpSSG RMS 2005 chemotherapy regimen, combined with an increase in the application of adequate local therapy, improvements in imaging and supportive care and potentially favourable patients' characteristics may have contributed to these results.

Sections du résumé

BACKGROUND
Infants (<12 months) with rhabdomyosarcoma have historically had poorer outcome than the older age groups. We present outcomes for infants and young children aged 12-36 months with localised rhabdomyosarcoma with a particular emphasis on infants.
PATIENTS AND METHODS
All children less than 36 months of age enrolled on the EpSSG RMS 2005 study for localised disease are included. Treatment comprised chemotherapy, local surgery and/or radiation therapy adapted to risk group and age. Main outcome measures were event free survival (EFS) and overall survival (OS).
RESULTS
Outcome data were available for 485/490 patients aged less than 36 months, 110 were infants. Infants received chemotherapy according to the risk group with no toxic deaths. Radiotherapy was delivered to 33.6% of infants and 63.5% of 12-36 months old, with respectively 41.7% and 22.2% receiving brachytherapy. Radical surgery was performed in 62% of infants and 57.1% of 12-36 months old. Median follow up for patients who are alive (n = 393) was 72.7 months (range 6.9-158.2). Five-year OS for infants was 88.4% (95%CI 80.3-93.2), which is significantly better than the OS in 12-36 months old patients of 78.0% (95%CI 73.2-82.0; p = 0.0204). Five-year EFS for infants was 72.5% (95%CI 62.8-80.0) compared with 66.1% (95%CI 61.0-70.7; p = 0.2663) for 12-36 months old.
CONCLUSION
Infants treated on RMS 2005 achieved excellent EFS and OS. The EpSSG RMS 2005 chemotherapy regimen, combined with an increase in the application of adequate local therapy, improvements in imaging and supportive care and potentially favourable patients' characteristics may have contributed to these results.

Identifiants

pubmed: 34865946
pii: S0959-8049(21)01189-8
doi: 10.1016/j.ejca.2021.10.031
pii:
doi:

Types de publication

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

Langues

eng

Sous-ensembles de citation

IM

Pagination

206-214

Subventions

Organisme : Cancer Research UK
ID : 28990
Pays : United Kingdom
Organisme : Cancer Research UK
ID : 5943
Pays : United Kingdom

Informations de copyright

Copyright © 2021 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

Olga Slater (O)

Department of Oncology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK. Electronic address: olga.slater@gosh.nhs.uk.

Jennifer E Gains (JE)

University College London Hospital NHS Foundation Trust, London, UK.

Anna M Kelsey (AM)

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

Federica De Corti (F)

University of Padova, Paediatric Surgery Women's and Health Department, Padova, Italy.

Ilaria Zanetti (I)

Istituto Oncologico Veneto IOV - IRCCS, Clinical Research Unit, Padua, Italy.

Beatrice Coppadoro (B)

Istituto Oncologico Veneto IOV - IRCCS, Clinical Research Unit, Padua, Italy.

Mette Jorgensen (M)

Department of Oncology, Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.

Soledad Gallego (S)

Pediatric Oncology, Hospital Universitari Vall d'Hebron, Barcelona, Spain.

Daniel H Orbach (DH)

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

Heidi Glosli (H)

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

Maja Cesen (M)

University Hospital Ljubljana, Department of Paediatric Haematology and Oncology, Ljubljana, Slovenia.

Mark N Gaze (MN)

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

Naima Smeulders (N)

Department of Urology, Great Ormond Street Hospital NHS Foundation Trust, London, UK.

Andrea Ferrari (A)

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

Meriel Jenney (M)

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

Veronique Minard-Colin (V)

Department of Pediatric and Adolescent Oncology, INSERM U1015, Gustave Roussy, Université Paris-Saclay, Villejuif, France.

Gianni Bisogno (G)

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

Johannes H M Merks (JHM)

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

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Classifications MeSH