Platinum-Based Regimens Are Active in Advanced Pediatric-Type Rhabdomyosarcoma in Adults and Depending on HMGB1 Expression

Int J Mol Sci. 2023 Jan 3;24(1):856. doi: 10.3390/ijms24010856.

Abstract

Rhabdomyosarcoma (RMS) in adults is a rare and aggressive disease, which lacks standard therapies for relapsed or advanced disease. This retrospective study aimed to describe the activity of BOMP-EPI (bleomycin, vincristine, methotrexate and cisplatin alternating with etoposide, cisplatin and ifosfamide), an alternative platinum-based regimen, in adult patients with relapsed/metastatic RMS. In the study, 10 patients with RMS with a median age at diagnosis of 20.8 years and a female/male distribution of 6/4 received a mean of 2.5 cycles of BOMP-EPI. The best RECIST response was a complete response in 1/10 (10%) patients, a partial response in 5/10 (50%), stable disease in 3/10 (30%) and progression in 1/10 (10%). With a median follow-up in the alive patients from the start of therapy of 30.5 months (15.7-258), all patients progressed with a median progression-free survival of 8.47 months (95% CI 8.1-8.8), and 7/10 patients died with a median overall survival of 24.7 months (95% CI 13.7-35.6). BOMP-EPI was an active chemotherapy regimen in adults with pediatric-type metastatic RMS, with outcomes in terms of survival that seem superior to what was expected for this poor-prognosis population. Low HMGB1 expression level was identified as a predictive factor of better response to this treatment.

Keywords: BOMP-EPI; HMGA2; HMGB1; HMGB2; cisplatin; rhabdomyosarcoma.

MeSH terms

  • Adult
  • Antineoplastic Combined Chemotherapy Protocols* / therapeutic use
  • Bleomycin
  • Child
  • Cisplatin / therapeutic use
  • Etoposide / therapeutic use
  • Female
  • HMGB1 Protein* / metabolism
  • Humans
  • Ifosfamide
  • Male
  • Mitomycin
  • Neoplasm Recurrence, Local / drug therapy
  • Retrospective Studies
  • Rhabdomyosarcoma / drug therapy
  • Rhabdomyosarcoma / pathology
  • Rhabdomyosarcoma, Embryonal* / drug therapy
  • Rhabdomyosarcoma, Embryonal* / metabolism
  • Vincristine / therapeutic use

Substances

  • Cisplatin
  • Etoposide
  • HMGB1 Protein
  • Vincristine

Supplementary concepts

  • BOMP protocol
  • ICE protocol 1

Grants and funding

This research was funded by IVAN PEREZ grant, GEIS number: GEISBECA2018. D.S.M. received a Sara Borrell postdoctoral fellowship funded by the National Institute of Health Carlos III (ISCIII) (CD20/00155).