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      Long-term disease control by trabectedin in a patient with dedifferentiated liposarcoma : A case report

      case-report
      , MD, , MD, PhD , , MD, PhD, , MD, PhD, , MD, PhD, , MD, PhD
      Medicine
      Wolters Kluwer Health
      liposarcoma, soft tissue sarcoma, trabectedin

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          Abstract

          Rationale:

          Trabectedin is an antitumor drug considered to be effective for liposarcoma, leiomyosarcoma, and translocation-related sarcoma. Concerning liposarcoma subtypes, evidence of the efficacy of trabectedin for dedifferentiated liposarcoma (DDLPS) is poor, whereas it is known to have high efficacy against myxoid liposarcoma. Moreover, there are few reports of long-term trabectedin treatment of cases of DDLPS. Here, we present a case of advanced metastatic DDLPS that achieved long-term disease control by trabectedin treatment.

          Patient concerns:

          A 68-year-old man presented with a mass in his back. Magnetic resonance imaging showed a tumorous mass in his back which exhibited low intensity on T1-weighted and high intensity on T2-weighted images.

          Diagnosis:

          The mass was diagnosed as DDLPS by open biopsy.

          Interventions:

          The patient underwent gemcitabine+docetaxel combination therapy followed by pazopanib and eribulin; all these therapies failed to halt disease progression. Subsequently, we changed the regimen to trabectedin as fourth-line therapy.

          Outcome:

          The patient achieved stable disease for approximately 18 months during 11 cycles of trabectedin therapy.

          Lessons:

          Trabectedin should be considered as a treatment option for DDLPS even in cases of numerous failed prior chemotherapy regimens.

          Related collections

          Most cited references6

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          Efficacy of trabectedin (ecteinascidin-743) in advanced pretreated myxoid liposarcomas: a retrospective study.

          Previous studies have suggested that trabectedin (ecteinascidin-743) could have antitumour activity in soft-tissue sarcoma. We aimed to study the usefulness of trabectedin in the treatment of patients with myxoid liposarcomas, a subtype of liposarcoma that is associated with specific chromosomal translocations t(12;16)(q13;p11) or t(12;22)(q13;q12) that result in the formation of DDIT3-FUS or DDIT3-EWSR1 fusion proteins. 51 patients with advanced pretreated myxoid liposarcoma who started treatment with trabectedin between April 4, 2001, and Sept 18, 2006 at five institutions in a compassionate-use programme were analysed retrospectively. Centralised radiological and pathological reviews were done for most patients. Trabectedin was given either as a 24-h continuous infusion or as a 3-h infusion, every 21 days, at 1.1-1.5 mg(2). 558 courses of trabectedin were given in total, with a median of ten courses for each patient (range 1-23). The primary endpoints were response rate and progression-free survival, and the secondary endpoint was overall survival. According to Response Evaluation Criteria in Solid Tumors (RECIST), after a median follow-up of 14.0 months (IQR 8.7-20.0), two patients had complete responses (CR) and 24 patients had partial responses (PR); the overall response was 51% (95% CI 36-65). Five patients had early progressive disease. In 17 of the 23 patients who achieved PR or CR as defined by RECIST and who had centralised radiological review, tissue-density changes, consisting of a decrease in tumour density on CT scan or a decrease in contrast enhancement on MRI (or both), preceded tumour shrinkage. Median progression-free survival was 14.0 months (13.1-21.0), and progression-free survival at 6 months was 88% (79-95). Trabectedin was associated with antitumour activity in this series of patients with myxoid liposarcoma. The noted patterns of tumour response were such that tissue density changes occurred before tumour shrinkage in several patients. In some patients, tissue-density changes only were seen. Long-lasting tumour control was noted in responsive patients. The compassionate-use programme is still ongoing. This analysis has resulted in the initiation of two prospective studies to assess the role of trabectedin in the treatment of patients with myxoid liposarcoma in preoperative and metastatic settings. Furthermore, the selective mechanism of action for trabectedin in this translocation-related sarcoma is being studied.
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            Clinical outcomes and safety with trabectedin therapy in patients with advanced soft tissue sarcomas following failure of prior chemotherapy: results of a worldwide expanded access program study.

            This expanded access program (EAP) was designed to provide trabectedin access for patients with incurable soft tissue sarcoma (STS) following progression of disease with standard therapy. The outcomes of trial participants accrued over approximately 5 years are reported.
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              Randomised phase III trial of trabectedin versus doxorubicin-based chemotherapy as first-line therapy in translocation-related sarcomas.

              This randomised phase III trial evaluated first-line trabectedin versus doxorubicin-based chemotherapy (DXCT) in patients with advanced/metastatic translocation-related sarcomas (TRS).
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                Author and article information

                Journal
                Medicine (Baltimore)
                Medicine (Baltimore)
                MEDI
                Medicine
                Wolters Kluwer Health
                0025-7974
                1536-5964
                January 2020
                10 January 2020
                : 99
                : 2
                : e18689
                Affiliations
                Department of Orthopaedic Surgery, Faculty of Medicine, Oita University, Oita, Japan.
                Author notes
                []Correspondence: Kazuhiro Tanaka, Department of Orthopaedic Surgery, Faculty of Medicine, Oita University, 1-1 Idaigaoka, Hasama, Yufu City, Oita 879-5593, Japan (e-mail: ktanaka@ 123456oita-u.ac.jp ).
                Article
                MD-D-19-03350 18689
                10.1097/MD.0000000000018689
                6959950
                31914068
                59e0fe9a-9cf4-46dc-b360-772ecb2a0412
                Copyright © 2020 the Author(s). Published by Wolters Kluwer Health, Inc.

                This is an open access article distributed under the Creative Commons Attribution License 4.0 (CCBY), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited. http://creativecommons.org/licenses/by/4.0

                History
                : 30 April 2019
                : 11 November 2019
                : 10 December 2019
                Categories
                5700
                Research Article
                Clinical Case Report
                Custom metadata
                TRUE

                liposarcoma,soft tissue sarcoma,trabectedin
                liposarcoma, soft tissue sarcoma, trabectedin

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