Ewing Sarcoma in Infants and Children Under 2 Years of Age: A French Retrospective Study

Ewing sarcoma, the second most common primary bone cancer in children, requires intensive treatment that may lead to significant long-term sequelae, particularly in infants. We retrospectively analyzed data from 1621 French patients treated between 1988 and 2015 within the EW88/93/97 or EE99 trials, focusing on 17 infants diagnosed before 24 months of age (1% of the cohort). Their outcomes were compared to the 1245 older pediatric patients (aged 2-18 years). Infant Ewing sarcomas were more frequently extraosseous (29% vs. 8%, p = 0.0037). Acute chemotherapy tolerance was comparable between infants and children under 2 years of age and older children. With a median follow-up of 13.2 years (range: 7.8-15), adapted chemotherapy doses and reduced use of radiotherapy for local treatment, outcomes in infants were favorable, with the same rate of 88% for 5-year disease-free survival (DFS) and overall survival (OS). Two infants experienced localized recurrences, one of whom had undergone incomplete tumor resection without additional local treatment. At the last follow-up, long-term side effects were predominantly associated with local treatments, particularly surgery, rather than chemotherapy. However, given the young age of the patients, certain late toxicities (e.g., fertility impairment) remain unevaluated or could manifest in later years. Adapting chemotherapy doses to age and weight, combined with optimal local treatment, enables high survival rates in infants with Ewing sarcoma. Further monitoring is essential to assess long-term sequelae as this population ages.

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Journal
Pediatric Blood & Cancer
Published
2026-09-10
DOI
https://doi.org/10.1002/1545-5017.70632
Primary Topic
Sarcoma Diagnosis and Treatment
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article
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article

Ewing Sarcoma in Infants and Children Under 2 Years of Age: A French Retrospective Study

Natacha Entz‐Werlé, Estelle Thébaud, Laure Saumet, Cyril Lervat et al.
Pediatric Blood & Cancer
Sarcoma Diagnosis and Treatment
article

Ewing Sarcoma in Infants and Children Under 2 Years of Age: A French Retrospective Study

Natacha Entz‐Werlé, Estelle Thébaud, Laure Saumet, Cyril Lervat, Marie‐Dominique Tabone, Nathalie Cozic, Élodie Verdier, Nathalie Gaspar, Sophie Taque, Gabriel Revon‐Rivière, Cécile Verite, Marta Jimenez, Gaëlle Pierron, Marie‐Pierre Castex, Stéphanie Proust, Dominique Plantaz, Gregory Guimard, Perrine Marec‐Berard, Sarah Winter
article en

Abstract

Ewing sarcoma, the second most common primary bone cancer in children, requires intensive treatment that may lead to significant long-term sequelae, particularly in infants. We retrospectively analyzed data from 1621 French patients treated between 1988 and 2015 within the EW88/93/97 or EE99 trials, focusing on 17 infants diagnosed before 24 months of age (1% of the cohort). Their outcomes were compared to the 1245 older pediatric patients (aged 2-18 years). Infant Ewing sarcomas were more frequently extraosseous (29% vs. 8%, p = 0.0037). Acute chemotherapy tolerance was comparable between infants and children under 2 years of age and older children. With a median follow-up of 13.2 years (range: 7.8-15), adapted chemotherapy doses and reduced use of radiotherapy for local treatment, outcomes in infants were favorable, with the same rate of 88% for 5-year disease-free survival (DFS) and overall survival (OS). Two infants experienced localized recurrences, one of whom had undergone incomplete tumor resection without additional local treatment. At the last follow-up, long-term side effects were predominantly associated with local treatments, particularly surgery, rather than chemotherapy. However, given the young age of the patients, certain late toxicities (e.g., fertility impairment) remain unevaluated or could manifest in later years. Adapting chemotherapy doses to age and weight, combined with optimal local treatment, enables high survival rates in infants with Ewing sarcoma. Further monitoring is essential to assess long-term sequelae as this population ages.

Pediatric Blood & Cancer
Centre National de la Recherche Scientifique (FR), Inserm (FR), Université de Montpellier (FR), Université Paris-Saclay (FR), Institut Gustave Roussy (FR), Centre Hospitalier Universitaire de Nantes (FR), Université Rennes 2 (FR), Sorbonne Université (FR), Hôpital Armand-Trousseau (FR), Assistance Publique Hôpitaux de Marseille (FR), Institut d’Hématologie et d’Oncologie Pédiatrique (FR), Laboratoire de Biophotonique et Pharmacologie (FR), Centre Hospitalier Universitaire de Montpellier (FR), Centre Hospitalier Universitaire de Reims (FR), Bordeaux Population Health (FR), Children's Hospital 2 (VN), Centre Oscar Lambret (FR), UniCancer Group (FR), Agir Pour les Maladies Chroniques (FR), Hôpital de la Timone (FR), Hôpital Civil, Strasbourg (FR), Université d'Angers (FR), Université de Rennes (FR), Université de Strasbourg (FR), Institut Curie (FR), Université Grenoble Alpes (FR), Université de Reims Champagne-Ardenne (FR), Nantes Université (FR)
Good health and well-being
Openalex Percentile: Top 11%
Sarcoma Diagnosis and Treatment
DOIOpen Access PDFOpenAlex
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