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Pazopanib maintenance therapy after tandem high-dose chemotherapy for disseminated Ewing sarcoma

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Abstract

The dismal prognosis of patients with disseminated Ewing sarcoma necessitates the development of novel treatment strategies. Pazopanib is an oral multi-targeted tyrosine kinase inhibitor that is active against advanced soft tissue sarcoma. However, the clinical activity and feasibility of pazopanib for treating Ewing sarcoma remain poorly understood. Moreover, clinical information on the use of tandem high-dose chemotherapy for Ewing sarcoma is limited. A 14-year-old boy with Ewing sarcoma was transferred to our hospital for treatment. Magnetic resonance imaging, computed tomography scans, and bone scintigraphy revealed multiple lesions in the pubis, ilium, ischium, femur, rib, cranial bone, thoracic vertebrae, sacrum, obturator muscle, adductor magnus muscle, testicular cord, and lungs. Bone scintigraphy after intensive chemotherapies confirmed that multiple abnormal accumulations were still present in the cranial bone and pubis. Subsequently, the patient received tandem high-dose chemotherapy including topotecan, and radiotherapy. Abnormal accumulations have disappeared in bone scintigraphy. Subsequently, pazopanib maintenance therapy was initiated. Despite the presence of innumerable lesions at diagnosis, the patient has been in near-complete remission for the past 1 year with pazopanib administration. This confirms that adding pazopanib maintenance therapy after tandem high-dose chemotherapy is a therapeutic option for cases with disseminated Ewing sarcoma.

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References

  1. Ladenstein R, Pötschger U, Le Deley MC et al (2010) Primary disseminated multifocal Ewing sarcoma: results of the Euro-EWING 99 trial. J Clin Oncol 28:3284–3291

    Article  CAS  PubMed  Google Scholar 

  2. Gaspar N, Hawkins DS, Dirksen U et al (2015) Ewing sarcoma: current management and future approaches through collaboration. J Clin Oncol 33:3036–3046

    Article  CAS  PubMed  Google Scholar 

  3. Paulussen M, Ahrens S, Burdach S et al (1998) Primary metastatic (stage IV) Ewing tumor: survival analysis of 171 patients from the EICESS studies. European Intergroup Cooperative Ewing Sarcoma Studies. Ann Oncol 9:275–281

    Article  CAS  PubMed  Google Scholar 

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

    Article  CAS  Google Scholar 

  5. Thiel U, Wawer A, von Luettichau I et al (2016) Bone marrow involvement identifies a subgroup of advanced Ewing sarcoma patients with fatal outcome irrespective of therapy in contrast to curable patients with multiple bone metastases but unaffected marrow. Oncotarget 7:70959–70968

    Article  PubMed  PubMed Central  Google Scholar 

  6. van der Graaf WT, Blay JY, Chawla SP et al (2012) Pazopanib for metastatic soft-tissue sarcoma (PALETTE): a randomised, double-blind, placebo-controlled phase 3 trial. Lancet 379:1879–1886

    Article  CAS  PubMed  Google Scholar 

  7. Juergens C, Weston C, Lewis I et al (2006) Safety assessment of intensive induction with vincristine, ifosfamide, doxorubicin, and etoposide (VIDE) in the treatment of Ewing tumors in the EURO-E.W.I.N.G. 99 clinical trial. Pediatr Blood Cancer 47:22–29

    Article  PubMed  Google Scholar 

  8. Casey DA, Wexler LH, Merchant MS et al (2009) Irinotecan and temozolomide for Ewing sarcoma: the Memorial Sloan-Kettering experience. Pediatr Blood Cancer 53:1029–1034

    Article  PubMed  Google Scholar 

  9. Burke MJ, Walterhouse DO, Jacobsohn DA et al (2007) Tandem high-dose chemotherapy with autologous peripheral hematopoietic progenitor cell rescue as consolidation therapy for patients with high-risk Ewing family tumors. Pediatr Blood Cancer 49:196–198

    Article  PubMed  Google Scholar 

  10. Donato ML, Gershenson DM, Wharton JT et al (2001) High-dose topotecan, melphalan, and cyclophosphamide (TMC) with stem cell support: a new regimen for the treatment of advanced ovarian cancer. Gynecol Oncol 82:420–426

    Article  CAS  PubMed  Google Scholar 

  11. Kazmi SM, Saliba RM, Donato M et al (2011) Phase II trial of high-dose topotecan, melphalan and CY with autologous stem cell support for multiple myeloma. Bone Marrow Transpl 46:510–515

    Article  CAS  Google Scholar 

  12. Yamamoto Y, Nozawa M, Shimizu N et al (2014) Pazopanib for recurrent extraosseous Ewing’s sarcoma of the retroperitoneum. Int J Urol 21:1183–1184

    Article  PubMed  Google Scholar 

  13. Attia S, Okuno SH, Robinson SI et al (2015) Clinical activity of pazopanib in metastatic extraosseous Ewing sarcoma. Rare Tumors 7:5992

    Article  PubMed  PubMed Central  Google Scholar 

  14. Alcindor T (2015) Response of refractory Ewing sarcoma to pazopanib. Acta Oncol 54:1063–1064

    Article  PubMed  Google Scholar 

  15. Mori Y, Kinoshita S, Kanamori T et al (2018) The successful treatment of metastatic extraosseous ewing sarcoma with pazopanib. Intern Med 57:2753–2757

    Article  PubMed  PubMed Central  Google Scholar 

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Correspondence to Akihiro Tamura.

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The authors have no conflict of interest to disclose. This study was conducted in accordance with the Declaration of Helsinki. Written and oral informed consent for publication was obtained from the patient’s guardians.

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Tamura, A., Yamamoto, N., Nino, N. et al. Pazopanib maintenance therapy after tandem high-dose chemotherapy for disseminated Ewing sarcoma. Int Canc Conf J 8, 95–100 (2019). https://doi.org/10.1007/s13691-019-00362-w

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  • DOI: https://doi.org/10.1007/s13691-019-00362-w

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