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Cancer Rep (Hoboken). 2021 Jun;4(3):e1329. doi: 10.1002/cnr2.1329. Epub 2021 Jan 16.

Clinical outcome of patients with recurrent or refractory localized Ewing's sarcoma family of tumors: A retrospective report from the Japan Ewing Sarcoma Study Group.

Cancer reports (Hoboken, N.J.)

Katsutsugu Umeda, Takako Miyamura, Kenji Yamada, Hideki Sano, Ako Hosono, Minako Sumi, Hajime Okita, Tadashi Kumamoto, Akira Kawai, Junya Hirayama, Ryoji Jyoko, Akihisa Sawada, Hideki Nakayama, Yosuke Hosoya, Naoko Maeda, Nobuyuki Yamamoto, Chihaya Imai, Daiichiro Hasegawa, Motoaki Chin, Toshifumi Ozaki,

Affiliations

  1. Department of Pediatrics, Graduate School of Medicine, Kyoto University, Kyoto, Japan.
  2. Department of Pediatrics, Osaka University Graduate School of Medicine, Suita, Japan.
  3. Department of Orthopedic Surgery, Okazaki City Hospital, Okazaki, Japan.
  4. Department of Pediatric Oncology, National Cancer Center Hospital East, Kashiwa, Japan.
  5. Department of Pediatric Oncology, Fukushima Medical University, Fukushima, Japan.
  6. Department of Radiation Oncology, Tokyo Metropolitan Geriatric Hospital, Tokyo, Japan.
  7. Department of Pathology, Keio University School of Medicine, Tokyo, Japan.
  8. Department of Pediatric Oncology, National Cancer Center Hospital, Tokyo, Japan.
  9. Musculoskeletal Oncology, National Cancer Center Hospital, Tokyo, Japan.
  10. Department of Pediatrics, Mie University Graduate School of Medicine, Tsu, Japan.
  11. Department of Orthopedic Surgery, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama, Japan.
  12. Department of Hematology/Oncology, Osaka Women's and Children's Hospital, Izumi, Japan.
  13. Department of Pediatrics, National Hospital Organization, Kyusyu Cancer Center, Fukuoka, Japan.
  14. Department of Pediatrics, St. Luke's International Hospital, Tokyo, Japan.
  15. Department of Pediatrics, National Hospital Organization Nagoya Medical Center, Nagoya, Japan.
  16. Department of Pediatrics, Kobe University Graduate School of Medicine, Kobe, Japan.
  17. Department of Pediatrics, Niigata University Graduate School of Medicine and Dental Sciences, Niigata, Japan.
  18. Department of Hematology and Oncology, Children's Cancer Center, Kobe Children's Hospital, Kobe, Japan.
  19. Department of Pediatrics and Child Health, Nihon University Itabashi Hospital, Tokyo, Japan.

PMID: 33452866 PMCID: PMC8222563 DOI: 10.1002/cnr2.1329

Abstract

BACKGROUND: Patients with Ewing's sarcoma family of tumors (ESFT) who experience relapse or progression have a poor prognosis.

AIM: This study aimed to identify the prognostic and therapeutic factors affecting overall survival (OS) of patients with recurrent or refractory localized ESFT.

METHODS AND RESULTS: Thirty-eight patients with localized ESFT who experienced first relapse or progression between 2000 and 2018 were retrospectively reviewed. The 5-year OS rate of the entire cohort was 48.3% (95% confidence interval, 29.9%-64.5%). Multivariate analysis of OS identified time to relapse or progression, but not stem cell transplantation (SCT), as the sole independent risk factor (hazard ratio, 35.8; P = .002). Among 31 patients who received salvage chemotherapy before local treatment, 21 received chemotherapy regimens that are not conventionally used for newly diagnosed ESFT. The objective response rate to first-line salvage chemotherapy was 55.2% in the 29 evaluable patients. Time to relapse or progression was significantly associated with response to first-line salvage chemotherapy (P = .006).

CONCLUSIONS: The present study fails to demonstrate significant clinical benefit of SCT for recurrent or refractory localized ESFT. Recently established chemotherapy regimens may increase the survival rate of patients with recurrent or refractory localized ESFT while attenuating the beneficial effect of SCT.

© 2020 The Authors. Cancer Reports published by Wiley Periodicals LLC.

Keywords: Ewing's sarcoma family of tumors; chemotherapy; progression; relapse; stem cell transplantation

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