Case Report: Response to Regional Melphalan via Limb Infusion and Systemic PD1 Blockade in Recurrent Myxofibrosarcoma: A Report of 2 Cases

Treatment options for patients with advanced sarcoma remain limited. Promising responses to checkpoint inhibition have been observed, but responses to single-agent PD-1 inhibition are rare. We report on two patients with multiply recurrent myxofibrosarcoma treated with the combination of regionally...

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Autores principales: Bartlett, Edmund K., D’Angelo, Sandra P., Kelly, Ciara M., Siegelbaum, Robert H., Fisher, Charles, Antonescu, Cristina R., Ariyan, Charlotte E.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8554327/
https://www.ncbi.nlm.nih.gov/pubmed/34722269
http://dx.doi.org/10.3389/fonc.2021.725484
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author Bartlett, Edmund K.
D’Angelo, Sandra P.
Kelly, Ciara M.
Siegelbaum, Robert H.
Fisher, Charles
Antonescu, Cristina R.
Ariyan, Charlotte E.
author_facet Bartlett, Edmund K.
D’Angelo, Sandra P.
Kelly, Ciara M.
Siegelbaum, Robert H.
Fisher, Charles
Antonescu, Cristina R.
Ariyan, Charlotte E.
author_sort Bartlett, Edmund K.
collection PubMed
description Treatment options for patients with advanced sarcoma remain limited. Promising responses to checkpoint inhibition have been observed, but responses to single-agent PD-1 inhibition are rare. We report on two patients with multiply recurrent myxofibrosarcoma treated with the combination of regionally administered melphalan (via isolated limb infusion) and pembrolizumab. Both patients had recurrent disease after multiple surgical resections and radiation. Analysis of primary tumors demonstrated microsatellite stable tumors with few mutations. After combination treatment, one patient had a significant partial response of 6 months duration, the second patient had a complete response of 2 years duration. Post treatment biopsies demonstrated immune infiltration into the tumor. These promising responses in patients with multiply recurrent myxofibrosarcoma have prompted the development of an investigator-initiated clinical trial to formally study the combination of regional melphalan and pembrolizumab in a systematic fashion (NCT04332874).
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spelling pubmed-85543272021-10-30 Case Report: Response to Regional Melphalan via Limb Infusion and Systemic PD1 Blockade in Recurrent Myxofibrosarcoma: A Report of 2 Cases Bartlett, Edmund K. D’Angelo, Sandra P. Kelly, Ciara M. Siegelbaum, Robert H. Fisher, Charles Antonescu, Cristina R. Ariyan, Charlotte E. Front Oncol Oncology Treatment options for patients with advanced sarcoma remain limited. Promising responses to checkpoint inhibition have been observed, but responses to single-agent PD-1 inhibition are rare. We report on two patients with multiply recurrent myxofibrosarcoma treated with the combination of regionally administered melphalan (via isolated limb infusion) and pembrolizumab. Both patients had recurrent disease after multiple surgical resections and radiation. Analysis of primary tumors demonstrated microsatellite stable tumors with few mutations. After combination treatment, one patient had a significant partial response of 6 months duration, the second patient had a complete response of 2 years duration. Post treatment biopsies demonstrated immune infiltration into the tumor. These promising responses in patients with multiply recurrent myxofibrosarcoma have prompted the development of an investigator-initiated clinical trial to formally study the combination of regional melphalan and pembrolizumab in a systematic fashion (NCT04332874). Frontiers Media S.A. 2021-10-15 /pmc/articles/PMC8554327/ /pubmed/34722269 http://dx.doi.org/10.3389/fonc.2021.725484 Text en Copyright © 2021 Bartlett, D’Angelo, Kelly, Siegelbaum, Fisher, Antonescu and Ariyan https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Oncology
Bartlett, Edmund K.
D’Angelo, Sandra P.
Kelly, Ciara M.
Siegelbaum, Robert H.
Fisher, Charles
Antonescu, Cristina R.
Ariyan, Charlotte E.
Case Report: Response to Regional Melphalan via Limb Infusion and Systemic PD1 Blockade in Recurrent Myxofibrosarcoma: A Report of 2 Cases
title Case Report: Response to Regional Melphalan via Limb Infusion and Systemic PD1 Blockade in Recurrent Myxofibrosarcoma: A Report of 2 Cases
title_full Case Report: Response to Regional Melphalan via Limb Infusion and Systemic PD1 Blockade in Recurrent Myxofibrosarcoma: A Report of 2 Cases
title_fullStr Case Report: Response to Regional Melphalan via Limb Infusion and Systemic PD1 Blockade in Recurrent Myxofibrosarcoma: A Report of 2 Cases
title_full_unstemmed Case Report: Response to Regional Melphalan via Limb Infusion and Systemic PD1 Blockade in Recurrent Myxofibrosarcoma: A Report of 2 Cases
title_short Case Report: Response to Regional Melphalan via Limb Infusion and Systemic PD1 Blockade in Recurrent Myxofibrosarcoma: A Report of 2 Cases
title_sort case report: response to regional melphalan via limb infusion and systemic pd1 blockade in recurrent myxofibrosarcoma: a report of 2 cases
topic Oncology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8554327/
https://www.ncbi.nlm.nih.gov/pubmed/34722269
http://dx.doi.org/10.3389/fonc.2021.725484
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