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Ann Surg Oncol. 2021 Jul;28(7):3893-3901. doi: 10.1245/s10434-020-09306-8. Epub 2020 Nov 04.

Clinical Outcomes of Patients with Metastatic Solitary Fibrous Tumors: A Japanese Musculoskeletal Oncology Group (JMOG) Multiinstitutional Study.

Annals of surgical oncology

Hidetatsu Outani, Eisuke Kobayashi, Junji Wasa, Masato Saito, Satoshi Takenaka, Keiko Hayakawa, Makoto Endo, Akihiko Takeuchi, Hiroshi Kobayashi, Munehisa Kito, Takeshi Morii, Jungo Imanishi, Takafumi Ueda

Affiliations

  1. Department of Orthopedic Surgery, Osaka University Graduate School of Medicine, Suita, Osaka, Japan. [email protected].
  2. Department of Musculoskeletal Oncology and Rehabilitation, National Cancer Center Hospital, Tokyo, Japan.
  3. Department of Orthopedic Oncology, Shizuoka Cancer Center Hospital, Shizuoka, Japan.
  4. Department of Orthopedic Surgery, School of Medicine, Keio University, Tokyo, Japan.
  5. Department of Musculoskeletal Oncology, Osaka International Cancer Institute, Osaka, Japan.
  6. Department of Orthopedic Surgery, Cancer Institute Hosipital of Japanese Foundation for Cancer Research, Tokyo, Japan.
  7. Department of Orthopedic Surgery, Kyushu University, Fukuoka, Japan.
  8. Department of Orthopedic Surgery, Kanazawa University Graduate School of Medical Sciences, Kanazawa, Japan.
  9. Department of Orthopedic Surgery, The University of Tokyo Hospital, Tokyo, Japan.
  10. Department of Orthopedic Surgery, Shinshu University School of Medicine, Matsumoto, Japan.
  11. Department of Orthopedic Surgery, Kyorin University, Tokyo, Japan.
  12. Department of Orthopedic Oncology and Surgery, Saitama Medical University International Medical Center, Hidaka, Saitama, Japan.
  13. Department of Orthopedic Surgery, National Hospital Organization Osaka National Hospital, Osaka, Japan.

PMID: 33146837 DOI: 10.1245/s10434-020-09306-8

Abstract

BACKGROUND: Although the unpredictable malignant behavior of solitary fibrous tumors (SFTs) has been recognized, the clinical features and prognosis of metastatic SFTs have not been well documented due to the extreme rarity of these cases. The aim of this study is to investigate the clinical features, prognostic factors, and optimal management of patients with metastatic SFTs.

PATIENTS AND METHODS: Sixty patients with metastatic SFT were retrospectively reviewed. Univariate and multivariate analyses were performed to identify the factors associated with survival. Time to next treatment (TNT) was used to evaluate the effects of various chemotherapy regimens.

RESULTS: A total of 34 male and 26 female patients (median age 55 years, range, 23-87 years) were included in the study. The median follow-up period after metastasis was 32 months (range 1-126 months). Tumor location and local recurrence were correlated with late metastasis. The 3- and 5-year overall survival rates were 72.7% and 49.2%, respectively. Primary tumor location, number of metastases, and metastasectomy were significantly associated with survival. Metastasectomy was the only significant variable on multivariate analysis. The TNT was significantly different among the various regimens.

CONCLUSIONS: Patients with metastatic SFTs had relatively longer survival periods compared with those with other metastatic soft-tissue sarcomas. Tumor location and number of metastases was associated with survival. Surgical resection of the metastatic lesions offers the best chance of survival, however further studies are warranted to define patients who would benefit from metastasectomy, and the most effective chemotherapeutic regimen for patients with metastatic SFTs remains unknown.

References

  1. Enzinger FM, Smith BH. Hemangiopericytoma An analysis of 106 cases. Hum Path. 1976;7(1):61–82. - PubMed
  2. Pasquali S, Gronchi A, Strauss D, et al. Resectable extra-pleural and extra-meningeal solitary fibrous tumours: A multi-centre prognostic study. Eur J Surg Ooncol. 2016;42(7):1064–1070. - PubMed
  3. Demicco EG, Park MS, Araujo DM, et al. Solitary fibrous tumor: a clinicopathological study of 110 cases and proposed risk assessment model. ModPathol. 2012;25(9):1298–1306. - PubMed
  4. Demicco EG, Wagner MJ, Maki RG, et al. Risk assessment in solitary fibrous tumors: validation and refinement of a risk stratification model. Mod Pathol. 2017;30(10):1433–1442. - PubMed
  5. Salas S, Resseguier N, Blay JY, et al. Prediction of local and metastatic recurrence in solitary fibrous tumor: construction of a risk calculator in a multicenter cohort from the French Sarcoma Group (FSG) database. Ann Oncol. 2017;28(8):1979–1987. - PubMed
  6. Demicco EG, Griffin AM, Gladdy RA, et al. Comparison of published risk models for prediction of outcome in patients with extrameningeal solitary fibrous tumour. Histopathology. 2019;75(5):723–737. - PubMed
  7. van Houdt WJ, Westerveld CM, Vrijenhoek JE, et al. Prognosis of solitary fibrous tumors: a multicenter study. Annals of surgical oncology. 2013;20(13):4090–4095. - PubMed
  8. Gholami S, Cassidy MR, Kirane A, et al. Size and location are the most important risk factors for malignant behavior in resected solitary fibrous tumors. Ann Surg Oncol. 2017;24(13):3865–3871. - PubMed
  9. Gold JS, Antonescu CR, Hajdu C, et al. Clinicopathologic correlates of solitary fibrous tumors. Cancer. 2002;94(4):1057–1068. - PubMed
  10. Tan MC, Brennan MF, Kuk D, et al. Histology-based classification predicts pattern of recurrence and improves risk stratification in primary retroperitoneal sarcoma. Ann Surg. 2016; 263(3):593–600. - PubMed
  11. Kanda Y. Investigation of the freely available easy-to-use software ‘EZR’ for medical statistics. Bone Marrow Transplant. 2013;48(3):452–458. - PubMed
  12. Machado I, Nieto-Morales G, Cruz J, et al. Controversial issues in soft tissue solitary fibrous tumors: A pathological and molecular review. Pathol Intl. 2020;70(3):129–139. - PubMed
  13. Italiano A, Mathoulin-Pelissier S, Cesne AL, et al. Trends in survival for patients with metastatic soft-tissue sarcoma. Cancer. 2011;117(5):1049–1054. - PubMed
  14. Judson I, Verweij J, Gelderblom H, et al. Doxorubicin alone versus intensified doxorubicin plus ifosfamide for first-line treatment of advanced or metastatic soft-tissue sarcoma: a randomised controlled phase 3 trial. Lancet. Oncol. 2014;15(4):415–423. - PubMed
  15. Tap WD, Wagner AJ, Schöffski P, et al. Effect of doxorubicin plus olaratumab vs doxorubicin plus placebo on survival in patients with advanced soft tissue sarcomas: The ANNOUNCE randomized clinical trial. JAMA. 2020;323(13):1266–1276. - PubMed
  16. Wilky BA, Montgomery EA, Guzzetta AA, Ahuja N, Meyer CF. Extrathoracic location and “borderline” histology are associated with recurrence of solitary fibrous tumors after surgical resection. Ann Surg Oncol. 2013;20(13):4080–4089. - PubMed
  17. Yamada Y, Kohashi K, Kinoshita I, et al. Clinicopathological review of solitary fibrous tumors: dedifferentiation is a major cause of patient death. Virchows Arch. 2019;475(4):467–477. - PubMed
  18. Martin-Broto J, Stacchiotti S, Lopez-Pousa A, et al. Pazopanib for treatment of advanced malignant and dedifferentiated solitary fibrous tumour: a multicentre, single-arm, phase 2 trial. Lancet Oncol. 2019;20(1):134–144. - PubMed
  19. Casson AG, Putnam JB, Natarajan G, et al. Five-year survival after pulmonary metastasectomy for adult soft tissue sarcoma. Cancer. 1992;69(3):662–668. - PubMed
  20. Kim S, Ott HC, Wright CD, et al. Pulmonary resection of metastatic sarcoma: prognostic factors associated with improved outcomes. Ann ThoracSurg. 2011;92(5):1780–1786; discussion 1786–1787. - PubMed
  21. Ueda T, Uchida A, Kodama K, et al. Aggressive pulmonary metastasectomy for soft tissue sarcomas. Cancer. 1993;72(6):1919–1925. - PubMed
  22. Wigge S, Heissner K, Steger V, et al. Impact of surgery in patients with metastatic soft tissue sarcoma: A monocentric retrospective analysis. J Surg Oncol. 2018;118(1):167-176. - PubMed
  23. Mizuno T, Taniguchi T, Ishikawa Y, et al. Pulmonary metastasectomy for osteogenic and soft tissue sarcoma: who really benefits from surgical treatment? Eur J Cardiothoracic Surg. 2013;43(4):795–799. - PubMed
  24. Yamamoto Y, Kanzaki R, Kanou T, et al. Long-term outcomes and prognostic factors of pulmonary metastasectomy for osteosarcoma and soft tissue sarcoma. Int J Clin Oncol. 2019;24(7):863–870. - PubMed
  25. Krengli M, Cena T, Zilli T, et al. Radiotherapy in the treatment of extracranial hemangiopericytoma/solitary fibrous tumor: Study from the Rare Cancer Network. Radiother Oncol. 2020;144:114–120. - PubMed
  26. Haas RL, Walraven I, Lecointe-Artzner E, et al. Radiation therapy as sole management for solitary fibrous tumors (SFT): A retrospective study from the global sft initiative in collaboration with the sarcoma patients EuroNet. Int J Radiat Oncol Biol Phys 1. 2018;101(5):1226–1233. - PubMed
  27. Stacchiotti S, Libertini M, Negri T, et al. Response to chemotherapy of solitary fibrous tumour: a retrospective study. Eur J Cancer (Oxford, England : 1990) 2013;49(10):2376–2383. - PubMed
  28. Park MS, Ravi V, Conley A, et al. The role of chemotherapy in advanced solitary fibrous tumors: a retrospective analysis. Clin Sarcoma Res. 2013;3(1):7. - PubMed
  29. Khalifa J, Ouali M, Chaltiel L, et al. Efficacy of trabected in malignant solitary fibrous tumors: a retrospective analysis from the French Sarcoma Group. BMC Cancer. 2015;15:700. - PubMed
  30. Park MS, Patel SR, Ludwig JA, et al. Activity of temozolomide and bevacizumab in the treatment of locally advanced, recurrent, and metastatic hemangiopericytoma and malignant solitary fibrous tumor. Cancer. 2011;117(21):4939–4947. - PubMed
  31. Maruzzo M, Martin-Liberal J, Messiou C, et al. Pazopanib as first line treatment for solitary fibrous tumours: the Royal Marsden Hospital experience. Clin Sarcoma Res. 2015;5:5. - PubMed
  32. Liang C, Li L, Fraser CD, et al. The treatment patterns, efficacy, and safety of nab ((R))-paclitaxel for the treatment of metastatic breast cancer in the United States: results from health insurance claims analysis. BMC Cancer. 2015;15:1019. - PubMed
  33. Savina M, Le Cesne A, Blay JY, et al. Patterns of care and outcomes of patients with METAstatic soft tissue SARComa in a real-life setting: the METASARC observational study. BMC Med. 2017;15(1):78. - PubMed

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