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Melan-A/MART-1 immunity in a EWS-ATF1 translocated clear cell sarcoma patient treated with sunitinib: a case report

BACKGROUND: Clear cell sarcoma (CCS), initially named malignant melanoma of soft parts, is an aggressive soft tissue sarcoma (STS) that, due to MITF activation, shares with melanoma the expression of melanocyte differentiation antigens. CCS is poorly sensitive to chemotherapy. Multi-kinase inhibitor...

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Autores principales: Tazzari, Marcella, Palassini, Elena, Vergani, Barbara, Villa, Antonello, Rini, Francesca, Negri, Tiziana, Colombo, Chiara, Crippa, Flavio, Morosi, Carlo, Casali, Paolo G, Pilotti, Silvana, Stacchiotti, Silvia, Rivoltini, Licia, Castelli, Chiara
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2015
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4342079/
https://www.ncbi.nlm.nih.gov/pubmed/25880253
http://dx.doi.org/10.1186/s12885-015-1044-0
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author Tazzari, Marcella
Palassini, Elena
Vergani, Barbara
Villa, Antonello
Rini, Francesca
Negri, Tiziana
Colombo, Chiara
Crippa, Flavio
Morosi, Carlo
Casali, Paolo G
Pilotti, Silvana
Stacchiotti, Silvia
Rivoltini, Licia
Castelli, Chiara
author_facet Tazzari, Marcella
Palassini, Elena
Vergani, Barbara
Villa, Antonello
Rini, Francesca
Negri, Tiziana
Colombo, Chiara
Crippa, Flavio
Morosi, Carlo
Casali, Paolo G
Pilotti, Silvana
Stacchiotti, Silvia
Rivoltini, Licia
Castelli, Chiara
author_sort Tazzari, Marcella
collection PubMed
description BACKGROUND: Clear cell sarcoma (CCS), initially named malignant melanoma of soft parts, is an aggressive soft tissue sarcoma (STS) that, due to MITF activation, shares with melanoma the expression of melanocyte differentiation antigens. CCS is poorly sensitive to chemotherapy. Multi-kinase inhibitors have been used as therapeutic agents. In the case we report here, treatment with sunitinib induced a long-lasting clinical response that was associated with an immune activation directed against Melan-A/MART-1 antigen. CASE PRESENTATION: A 28 years old female patient with an advanced molecularly confirmed CCS resistant to conventional chemotherapy was started in January 2012 on sunitinib, 37.5 mg/day, with evidence of radiologic and metabolic response at the primary and metastatic sites of disease. Pathologic response and loss of the Melan-A/MART-1 antigen were evidenced on residual tumor removed in April 2012. Immunological monitoring performed on patient’s blood during pharmacological treatment revealed a systemic, Melan-A/MART-1 specific immunity and a low frequency of immunosuppressive cells. Sunitinib was restarted in May 2012, with a new response, and continued for 11 months although with repeatedly interruptions due to toxicity. Disease progression and new responses were documented at each treatment interruption and restart. Sunitinib was definitively interrupted in April 2013 for disease progression. CONCLUSION: The analysis of this case proves that antigens expressed by CCS, as for melanoma, can be immunogenic in vivo and that tumor-antigen specific T cells may exert anti-tumor activity in CCS patient. Thus, manipulation of the immune response may have therapeutic potential for this STS subtype and immunotherapy approaches, can be promising therapeutic options for these patients.
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spelling pubmed-43420792015-02-27 Melan-A/MART-1 immunity in a EWS-ATF1 translocated clear cell sarcoma patient treated with sunitinib: a case report Tazzari, Marcella Palassini, Elena Vergani, Barbara Villa, Antonello Rini, Francesca Negri, Tiziana Colombo, Chiara Crippa, Flavio Morosi, Carlo Casali, Paolo G Pilotti, Silvana Stacchiotti, Silvia Rivoltini, Licia Castelli, Chiara BMC Cancer Case Report BACKGROUND: Clear cell sarcoma (CCS), initially named malignant melanoma of soft parts, is an aggressive soft tissue sarcoma (STS) that, due to MITF activation, shares with melanoma the expression of melanocyte differentiation antigens. CCS is poorly sensitive to chemotherapy. Multi-kinase inhibitors have been used as therapeutic agents. In the case we report here, treatment with sunitinib induced a long-lasting clinical response that was associated with an immune activation directed against Melan-A/MART-1 antigen. CASE PRESENTATION: A 28 years old female patient with an advanced molecularly confirmed CCS resistant to conventional chemotherapy was started in January 2012 on sunitinib, 37.5 mg/day, with evidence of radiologic and metabolic response at the primary and metastatic sites of disease. Pathologic response and loss of the Melan-A/MART-1 antigen were evidenced on residual tumor removed in April 2012. Immunological monitoring performed on patient’s blood during pharmacological treatment revealed a systemic, Melan-A/MART-1 specific immunity and a low frequency of immunosuppressive cells. Sunitinib was restarted in May 2012, with a new response, and continued for 11 months although with repeatedly interruptions due to toxicity. Disease progression and new responses were documented at each treatment interruption and restart. Sunitinib was definitively interrupted in April 2013 for disease progression. CONCLUSION: The analysis of this case proves that antigens expressed by CCS, as for melanoma, can be immunogenic in vivo and that tumor-antigen specific T cells may exert anti-tumor activity in CCS patient. Thus, manipulation of the immune response may have therapeutic potential for this STS subtype and immunotherapy approaches, can be promising therapeutic options for these patients. BioMed Central 2015-02-14 /pmc/articles/PMC4342079/ /pubmed/25880253 http://dx.doi.org/10.1186/s12885-015-1044-0 Text en © Tazzari et al.; licensee BioMed Central. 2015 This article is published under license to BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Case Report
Tazzari, Marcella
Palassini, Elena
Vergani, Barbara
Villa, Antonello
Rini, Francesca
Negri, Tiziana
Colombo, Chiara
Crippa, Flavio
Morosi, Carlo
Casali, Paolo G
Pilotti, Silvana
Stacchiotti, Silvia
Rivoltini, Licia
Castelli, Chiara
Melan-A/MART-1 immunity in a EWS-ATF1 translocated clear cell sarcoma patient treated with sunitinib: a case report
title Melan-A/MART-1 immunity in a EWS-ATF1 translocated clear cell sarcoma patient treated with sunitinib: a case report
title_full Melan-A/MART-1 immunity in a EWS-ATF1 translocated clear cell sarcoma patient treated with sunitinib: a case report
title_fullStr Melan-A/MART-1 immunity in a EWS-ATF1 translocated clear cell sarcoma patient treated with sunitinib: a case report
title_full_unstemmed Melan-A/MART-1 immunity in a EWS-ATF1 translocated clear cell sarcoma patient treated with sunitinib: a case report
title_short Melan-A/MART-1 immunity in a EWS-ATF1 translocated clear cell sarcoma patient treated with sunitinib: a case report
title_sort melan-a/mart-1 immunity in a ews-atf1 translocated clear cell sarcoma patient treated with sunitinib: a case report
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4342079/
https://www.ncbi.nlm.nih.gov/pubmed/25880253
http://dx.doi.org/10.1186/s12885-015-1044-0
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