Objective Responses in Metastatic Pleomorphic Rhabdomyosarcoma Treated with Combination of Doxorubicin and Pembrolizumab: A Case Series

Ioannis Kournoutas; Varun Monga; Jonathan Davick; John Rieth

doi:10.1159/000535959

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Objective Responses in Metastatic Pleomorphic Rhabdomyosarcoma Treated with Combination of Doxorubicin and Pembrolizumab: A Case Series

Journal article

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Objective Responses in Metastatic Pleomorphic Rhabdomyosarcoma Treated with Combination of Doxorubicin and Pembrolizumab: A Case Series

Ioannis Kournoutas, Varun Monga, Jonathan Davick and John Rieth

Case reports in oncology, Vol.17(1), pp.344-351

01/2024

DOI: 10.1159/000535959

PMCID: PMC10890802

PMID: 38404403

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Abstract

Pleomorphic rhabdomyosarcoma is a rare subtype of rhabdomyosarcoma, a soft tissue sarcoma with skeletal muscle differentiation. Although rhabdomyosarcoma is typically seen in the pediatric population, the pleomorphic variant most frequently presents in adulthood and is characteristically aggressive with no currently established treatment regimen in the setting of metastatic disease. There has been growing interest in the application of immune checkpoint inhibitors alongside conventional chemotherapeutic agents in the treatment of pleomorphic rhabdomyosarcoma. In the present case series, we report 2 patients with metastatic pleomorphic rhabdomyosarcoma treated with combination doxorubicin and pembrolizumab who had confirmed objective responses. Of note, these 2 patients had variable PD-L1 status - negative and low positive. Duration of treatment response was notable at 14 months and 9 months, respectively, with the first patient remaining on maintenance pembrolizumab therapy and the second patient subsequently achieving complete response with third-line trabectedin. Both patients are currently undergoing routine interval imaging with no evidence of disease at this time. This report highlights and discusses the potential role of PD-1 blockade in the treatment of pleomorphic rhabdomyosarcoma and also discusses burgeoning immunological data that may explain the clinical responses seen in these 2 cases.

Pembrolizumab

Immune checkpoint

Rhabdomyosarcoma

Pleomorphic rhabdomyosarcoma

Case series

Doxorubicin

Details

Title: Subtitle: Objective Responses in Metastatic Pleomorphic Rhabdomyosarcoma Treated with Combination of Doxorubicin and Pembrolizumab: A Case Series
Creators: Ioannis Kournoutas - Department of Internal Medicine, University of Iowa Carver College of Medicine, Iowa City, IA, USA
Varun Monga - University of Iowa
Jonathan Davick - University of Iowa
John Rieth - University of Iowa
Resource Type: Journal article
Publication Details: Case reports in oncology, Vol.17(1), pp.344-351
DOI: 10.1159/000535959
PMID: 38404403
PMCID: PMC10890802
NLM abbreviation: Case Rep Oncol
ISSN: 1662-6575
eISSN: 1662-6575
Language: English
Date published: 01/2024
Academic Unit: Hematology, Oncology, and Blood & Marrow Transplantation; Pathology; Internal Medicine
Record Identifier: 9984563458702771

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