Remarkable response to anti-PD1 immunotherapy in refractory metastatic high-grade myxofibrosarcoma patient: A case report.
Adult
Antibodies, Monoclonal, Humanized
Antineoplastic Agents
/ administration & dosage
Dissection
/ methods
Humans
Immunotherapy
/ methods
Lung Neoplasms
/ immunology
Male
Myxosarcoma
/ pathology
Neoplasm Grading
Neoplasm Staging
Programmed Cell Death 1 Receptor
/ antagonists & inhibitors
Progression-Free Survival
Soft Tissue Neoplasms
/ pathology
Thigh
/ pathology
Treatment Outcome
Journal
Medicine
ISSN: 1536-5964
Titre abrégé: Medicine (Baltimore)
Pays: United States
ID NLM: 2985248R
Informations de publication
Date de publication:
26 Mar 2021
26 Mar 2021
Historique:
received:
30
12
2020
accepted:
04
03
2021
entrez:
25
3
2021
pubmed:
26
3
2021
medline:
7
4
2021
Statut:
ppublish
Résumé
Myxofibrosarcoma (MFS) is a locally aggressive tumor and has the potential to be fatal because of distant metastasis. Immunotherapy targeting either programmed cell death protein 1 (PD-1) or programmed death ligand 1 (PD-L1) has recently shown a curative effect on multiple cancers including melanoma, non-small cell lung cancer, and renal cell carcinoma. Although the immunotherapy has been applied in sarcoma, there is little information about the efficiency to treat metastatic MFS. A 42-year-old male presented to the clinic with a mass in the left thigh. Mass resection and ligament replacement surgery were performed. The patient was diagnosed as high-grade MFS (federation nationale des centres de lutte contre le cancer, Grade 3) with pulmonary metastasis. In the past few years, he was treated with surgery, chemoradiotherapy, and Anlotinib (an angiogenesis inhibitor), but the metastatic lesion continued to progress. About 40% to 50% of tumor cells in his pulmonary tissues were showed positive PD-L1 expression and his tumor mutational burden was 215Muts. Thus, he received Camrelizumab (PD-1 inhibitor). Six months after the initiating immunotherapy of Camrelizumab, the size of pulmonary lesions showed marked shrinkage, indicating a partial response. After a follow-up of 18 months, the patient remained in good condition without progressive disease. This case described here demonstrated that immunotherapy of PD-1 inhibitor is a promising treatment option for refractory MFS with PD-L1 positive or tumor mutational burden -high, which could contribute to effective tumor response.
Identifiants
pubmed: 33761725
doi: 10.1097/MD.0000000000025262
pii: 00005792-202103260-00101
pmc: PMC9281969
doi:
Substances chimiques
Antibodies, Monoclonal, Humanized
0
Antineoplastic Agents
0
Programmed Cell Death 1 Receptor
0
camrelizumab
73096E137E
Types de publication
Case Reports
Journal Article
Langues
eng
Sous-ensembles de citation
IM
Pagination
e25262Subventions
Organisme : Support Program for Science and Technology of Sichuan Province
ID : 2017SZ0106
Informations de copyright
Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.
Déclaration de conflit d'intérêts
The authors have no conflicts of interest to disclose.
Références
Roland CL, Wang WL, Lazar AJ, et al. Myxofibrosarcoma. Surg Oncol Clin N Am 2016;25:775–88.
Teurneau H, Engellau J, Ghanei I, et al. High recurrence rate of myxofibrosarcoma: the effect of radiotherapy is not clear. Sarcoma 2019;2019:8517371–18517371.
Nascimento AF, Bertoni F, Fletcher CD. Epithelioid variant of myxofibrosarcoma: expanding the clinicomorphologic spectrum of myxofibrosarcoma in a series of 17 cases. Am J Surg Pathol 2007;31:99–105.
Torabi A, Amaya CN, Wians FH, et al. Pd-1 and Pd-L1 expression in bone and soft tissue sarcomas. Pathology 2017;49:506–13.
Tamura K, Hasegawa K, Katsumata N, et al. Efficacy and safety of nivolumab in Japanese patients with uterine cervical cancer, uterine corpus cancer, or soft tissue sarcoma: multicenter, open-label phase 2 trial. Cancer Sci 2019;110:2894–904.
Tawbi HA, Burgess M, Bolejack V, et al. Pembrolizumab in advanced soft-tissue sarcoma and bone sarcoma (SARC028): a multicentre, two-cohort, single-arm, open-label, phase 2 trial. Lancet Oncol 2017;1811:1493–501.
Tsuchie H, Kaya M, Nagasawa H, et al. Distant metastasis in patients with myxofibrosarcoma. Upsala J Med Sci 2017;122:190–3.
Kikuta K, Nakayama R, Yoshida A, et al. A histological positive margin after surgery is correlated with high local re-recurrence rate in patients with recurrent myxofibrosarcoma. Jpn J Clin Oncol 2017;47:334–41.
McMillan RR, Sima CS, Moraco NH, et al. Recurrence patterns after resection of soft tissue sarcomas of the chest wall. Ann Thorac Surg 2013;96:1223–8.
Kosemehmetoglu K, Ozogul E, Babaoglu B, et al. Programmed death ligand 1 (PD-L1) expression in malignant mesenchymal tumors. Turk J Pathol 2017;33:192–7.
Budczies J, Mechtersheimer G, Denkert C, et al. PD-L1 (CD274) copy number gain, expression, and immune cell infiltration as candidate predictors for response to immune checkpoint inhibitors in soft-tissue sarcoma. Oncoimmunology 2017;6:e1279777.
Fukui T, Wakatsuki Y, Matsukura T. Immunotherapy for advanced lung cancer combined with surgery for mediastinal myxofibrosarcoma: a case report. Surg Case Rep 2019;5:37–9.
Zheng B, Qu Y, Wang J, et al. Pathogenic and targetable genetic alterations in resected recurrent undifferentiated pleomorphic sarcomas identified by targeted next-generation sequencing. Cancer Genom Proteom 2019;16:221–8.
Chalmers ZR, Connelly CF, Fabrizio D, et al. Analysis of 100,000 human cancer genomes reveals the landscape of tumor mutational burden. Genome Med 2017;9:34–48.
Song HN, Kang MG, Park JR, et al. Pembrolizumab for refractory metastatic myxofibrosarcoma: a case report. Cancer Res Treat 2018;50:1458–61.
D’Angelo SP, Mahoney MR, Van Tine BA, et al. Nivolumab with or without ipilimumab treatment for metastatic sarcoma (Alliance A091401): two open-label, non-comparative, randomised, phase 2 trials. Lancet Oncol 2018;19:416–26.