Angiosarcoma of bone: a retrospective study of the European Musculoskeletal Oncology Society (EMSOS) View Full Text


Ontology type: schema:ScholarlyArticle      Open Access: True


Article Info

DATE

2020-07-02

AUTHORS

Emanuela Palmerini, Andreas Leithner, Reinhard Windhager, Georg Gosheger, Kjetil Boye, Minna Laitinen, Jendrik Hardes, Frank Traub, Paul Jutte, Madeleine Willegger, Jose’ Casanova, Elisabetta Setola, Alberto Righi, Piero Picci, Davide Maria Donati, Stefano Ferrari

ABSTRACT

Angiosarcoma of bone (B-AS) is a rare malignant tumor of vascular origin. The aim of this retrospective study is to report on treatments and prognosis. Data were collected from the EMSOS website. 80 patients in 9 centers included: 51 male/29 female; median age 54 years (range 17 to 92); 56% with localized disease, 44% metastatic. Primary tumor surgery: 76% (30% amputation, 26% intralesional margins); radiotherapy (RT): 41%; chemotherapy (CT): 47% (56% in metastatic, 41% in localized cases). With a median follow-up of 31 months (range 40 to 309), 5-year overall survival (OS) was 27% (95%CI 16–30): 41% (95%CI 25–56) for localized patients, and 8% (95%CI 0–20) for metastatic (p = 0.002). In metastatic patients, 1 year OS was significantly influenced by chemotherapy response: 67% (95CI% 29–100) for those who responded or had stable disease (n = 7), and 18% (95CI% 0–41) for patients with progressive disease (n = 11), p 0.002. The surgical complete remission (SCR) status was pivotal in localized patients (5-year OS 45% for SCR, 17% no SCR, p = 0.03); also 5-year OS was significantly influenced by age and site of the tumor. After multivariate analysis, the addition of radiotherapy to surgery significantly influenced the disease-free survival (DFS) rate, whereas the use of chemotherapy lost the significance showed at the univariate analysis. Overall, patients with metastatic B-AS have a dismal prognosis, with a prolonged survival in case with a response to chemotherapy. Experimental trials with more active systemic treatment regimens are needed. In patients with localized disease, the patient’s age and site of the tumor are prognostic factors and any effort must be made to achieve an SCR status. No definitive conclusions can be drawn from our data on the use of adjuvant chemotherapy, while the use of adjuvant radiotherapy might improve DSF in patients surgically free of disease. More... »

PAGES

10853

Identifiers

URI

http://scigraph.springernature.com/pub.10.1038/s41598-020-66579-5

DOI

http://dx.doi.org/10.1038/s41598-020-66579-5

DIMENSIONS

https://app.dimensions.ai/details/publication/pub.1128916031

PUBMED

https://www.ncbi.nlm.nih.gov/pubmed/32616718


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27 schema:description Angiosarcoma of bone (B-AS) is a rare malignant tumor of vascular origin. The aim of this retrospective study is to report on treatments and prognosis. Data were collected from the EMSOS website. 80 patients in 9 centers included: 51 male/29 female; median age 54 years (range 17 to 92); 56% with localized disease, 44% metastatic. Primary tumor surgery: 76% (30% amputation, 26% intralesional margins); radiotherapy (RT): 41%; chemotherapy (CT): 47% (56% in metastatic, 41% in localized cases). With a median follow-up of 31 months (range 40 to 309), 5-year overall survival (OS) was 27% (95%CI 16–30): 41% (95%CI 25–56) for localized patients, and 8% (95%CI 0–20) for metastatic (p = 0.002). In metastatic patients, 1 year OS was significantly influenced by chemotherapy response: 67% (95CI% 29–100) for those who responded or had stable disease (n = 7), and 18% (95CI% 0–41) for patients with progressive disease (n = 11), p 0.002. The surgical complete remission (SCR) status was pivotal in localized patients (5-year OS 45% for SCR, 17% no SCR, p = 0.03); also 5-year OS was significantly influenced by age and site of the tumor. After multivariate analysis, the addition of radiotherapy to surgery significantly influenced the disease-free survival (DFS) rate, whereas the use of chemotherapy lost the significance showed at the univariate analysis. Overall, patients with metastatic B-AS have a dismal prognosis, with a prolonged survival in case with a response to chemotherapy. Experimental trials with more active systemic treatment regimens are needed. In patients with localized disease, the patient’s age and site of the tumor are prognostic factors and any effort must be made to achieve an SCR status. No definitive conclusions can be drawn from our data on the use of adjuvant chemotherapy, while the use of adjuvant radiotherapy might improve DSF in patients surgically free of disease.
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34 European Musculoskeletal Oncology Society
35 Oncology Society
36 addition
37 addition of radiotherapy
38 adjuvant chemotherapy
39 adjuvant radiotherapy
40 age
41 age 54 years
42 aim
43 analysis
44 angiosarcoma
45 angiosarcoma of bone
46 bone
47 cases
48 center
49 chemotherapy
50 chemotherapy response
51 complete remission status
52 conclusion
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54 definitive conclusions
55 disease
56 disease-free survival rates
57 dismal prognosis
58 efforts
59 experimental trials
60 factors
61 localized disease
62 malignant tumors
63 median age 54 years
64 metastatic
65 metastatic patients
66 months
67 multivariate analysis
68 origin
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70 patient age
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76 rare malignant tumor
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79 remission status
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83 sites
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85 stable disease
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