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Solid Cancers

Tandem high-dose chemotherapy strategy as first-line treatment of primary disseminated multifocal Ewing sarcomas in children, adolescents and young adults

Bone Marrow Transplantation volume 50, pages 1083–1088 (2015)Cite this article

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Abstract

The prognosis of primary disseminated multifocal metastatic Ewing’s sarcoma (PDMES) is poor even if a slight improvement has been achieved with high-dose alkylating agent-containing chemotherapy. To enhance treatment efficacy, we assessed the feasibility, safety and efficacy of a tandem high-dose chemotherapy (HDC) regimen. In a single institution, patients with PDMES received six courses of vincristine/ifosfamide/doxorubicin/etoposide induction therapy, followed by high-dose thiotepa, and then melphalan–busulfan, 8 weeks apart. Surgical resection of primary tumour was carried out between the two HDC regimens and 70 days after the last HDC regimen for post-operative radiotherapy or irradiation alone. From October 2002 to 2009, 13 of the 18 consecutive patients with PDMES (72%) received the full treatment programme. The other five patients experienced early progression and died. Among the 13 patients, 11 relapsed after the end of the treatment programme within 6 months (2.2–11.9) from end of therapy. Only two patients are still alive in first complete remission after 9 years. The 3-year event-free survival (EFS) and overall survival (OS) rates were 11 and 22%, respectively. The median EFS and OS duration from the diagnosis were 13.4 and 17.3 months, respectively. Neither major complications nor treatment-related death occurred. The tandem-HDC regimen was feasible, with expected side effects, but it did not improve the outcome of patients with PDMES.

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Acknowledgements

The authors thank all the patients who participated in the study and the Association Enfants et Santé, Société Française de Lutte Contre les Cancers et les Leucémies de l’Enfant et de l’Adolescent (SFCE) for Research Support. They also thank L Saint-Ange for editing.

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Authors and Affiliations

  1. Département de cancérologie de l'enfant et de l'adolescent, Gustave Roussy, Villejuif, France

    S Loschi, C Dufour, O Oberlin, D Valteau-Couanet & N Gaspar

  2. Département de biostatistiques, Gustave Roussy, Villejuif, France

    G Goma

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  1. S Loschi
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Correspondence to N Gaspar.

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Loschi, S., Dufour, C., Oberlin, O. et al. Tandem high-dose chemotherapy strategy as first-line treatment of primary disseminated multifocal Ewing sarcomas in children, adolescents and young adults. Bone Marrow Transplant 50, 1083–1088 (2015). https://doi.org/10.1038/bmt.2015.118

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