Piotr Remiszewski, Sławomir Falkowski, Anna Szumera-Ciećkiewicz, Mateusz J Spałek, Piotr Rutkowski, Anna M Czarnecka
{"title":"从发病机制到患者床边:骨骼外黏液样软骨肉瘤的全面回顾。","authors":"Piotr Remiszewski, Sławomir Falkowski, Anna Szumera-Ciećkiewicz, Mateusz J Spałek, Piotr Rutkowski, Anna M Czarnecka","doi":"10.1007/s00432-025-06316-5","DOIUrl":null,"url":null,"abstract":"<p><p>Extraskeletal myxoid chondrosarcoma (EMC) is characterised by recurrent NR4A3 gene rearrangements, most commonly EWSR1::NR4A3, and accounts for approximately 1-3% of soft-tissue sarcomas (STS). It typically arises in the deep soft tissues of the proximal lower limb, particularly the thigh. Diagnosis is best established by integrating morphology and immunophenotype with molecular confirmation; in particular, NR4A3 break-apart fluorescence in situ hybridisation (FISH) provides a practical single-assay solution. For localised disease, complete surgical excision remains the cornerstone of treatment. Radiotherapy (RT) improves local control when margins are close or tumours are large. Recurrence-free survival (RFS) varies: local recurrence (LR) rates range from 13 to 42% across studies, and distant metastases develop in around 35-45% of patients, primarily in the lungs. The median time to metastasis is approximately 28 months. Overall survival (OS) reflects the typically indolent yet metastatic course: 5-year OS 66-88%, and 10-year disease-specific survival approximately 85%. In advanced disease, anthracycline-based chemotherapy yields a low objective response rate (ORR), although occasional partial responses occur. By contrast, the anti-angiogenic tyrosine kinase inhibitor pazopanib produced an ORR of 18% and a median progression-free survival (PFS) of 19 months in a multicentre phase 2 study (NCT02066285). No clinically validated agents directly target NR4A3. This review summarises contemporary diagnostics and treatment, emphasising high-quality surgery, selective RT, and consideration of anti-angiogenic tyrosine kinase inhibitors in advanced disease.</p>","PeriodicalId":15118,"journal":{"name":"Journal of Cancer Research and Clinical Oncology","volume":"151 11","pages":"283"},"PeriodicalIF":2.8000,"publicationDate":"2025-10-07","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12504171/pdf/","citationCount":"0","resultStr":"{\"title\":\"From pathogenesis to the patient's bedside: a comprehensive review of extraskeletal myxoid chondrosarcoma.\",\"authors\":\"Piotr Remiszewski, Sławomir Falkowski, Anna Szumera-Ciećkiewicz, Mateusz J Spałek, Piotr Rutkowski, Anna M Czarnecka\",\"doi\":\"10.1007/s00432-025-06316-5\",\"DOIUrl\":null,\"url\":null,\"abstract\":\"<p><p>Extraskeletal myxoid chondrosarcoma (EMC) is characterised by recurrent NR4A3 gene rearrangements, most commonly EWSR1::NR4A3, and accounts for approximately 1-3% of soft-tissue sarcomas (STS). It typically arises in the deep soft tissues of the proximal lower limb, particularly the thigh. Diagnosis is best established by integrating morphology and immunophenotype with molecular confirmation; in particular, NR4A3 break-apart fluorescence in situ hybridisation (FISH) provides a practical single-assay solution. For localised disease, complete surgical excision remains the cornerstone of treatment. Radiotherapy (RT) improves local control when margins are close or tumours are large. Recurrence-free survival (RFS) varies: local recurrence (LR) rates range from 13 to 42% across studies, and distant metastases develop in around 35-45% of patients, primarily in the lungs. The median time to metastasis is approximately 28 months. Overall survival (OS) reflects the typically indolent yet metastatic course: 5-year OS 66-88%, and 10-year disease-specific survival approximately 85%. In advanced disease, anthracycline-based chemotherapy yields a low objective response rate (ORR), although occasional partial responses occur. By contrast, the anti-angiogenic tyrosine kinase inhibitor pazopanib produced an ORR of 18% and a median progression-free survival (PFS) of 19 months in a multicentre phase 2 study (NCT02066285). No clinically validated agents directly target NR4A3. This review summarises contemporary diagnostics and treatment, emphasising high-quality surgery, selective RT, and consideration of anti-angiogenic tyrosine kinase inhibitors in advanced disease.</p>\",\"PeriodicalId\":15118,\"journal\":{\"name\":\"Journal of Cancer Research and Clinical Oncology\",\"volume\":\"151 11\",\"pages\":\"283\"},\"PeriodicalIF\":2.8000,\"publicationDate\":\"2025-10-07\",\"publicationTypes\":\"Journal Article\",\"fieldsOfStudy\":null,\"isOpenAccess\":false,\"openAccessPdf\":\"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC12504171/pdf/\",\"citationCount\":\"0\",\"resultStr\":null,\"platform\":\"Semanticscholar\",\"paperid\":null,\"PeriodicalName\":\"Journal of Cancer Research and Clinical Oncology\",\"FirstCategoryId\":\"3\",\"ListUrlMain\":\"https://doi.org/10.1007/s00432-025-06316-5\",\"RegionNum\":3,\"RegionCategory\":\"医学\",\"ArticlePicture\":[],\"TitleCN\":null,\"AbstractTextCN\":null,\"PMCID\":null,\"EPubDate\":\"\",\"PubModel\":\"\",\"JCR\":\"Q3\",\"JCRName\":\"ONCOLOGY\",\"Score\":null,\"Total\":0}","platform":"Semanticscholar","paperid":null,"PeriodicalName":"Journal of Cancer Research and Clinical Oncology","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1007/s00432-025-06316-5","RegionNum":3,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"","PubModel":"","JCR":"Q3","JCRName":"ONCOLOGY","Score":null,"Total":0}
From pathogenesis to the patient's bedside: a comprehensive review of extraskeletal myxoid chondrosarcoma.
Extraskeletal myxoid chondrosarcoma (EMC) is characterised by recurrent NR4A3 gene rearrangements, most commonly EWSR1::NR4A3, and accounts for approximately 1-3% of soft-tissue sarcomas (STS). It typically arises in the deep soft tissues of the proximal lower limb, particularly the thigh. Diagnosis is best established by integrating morphology and immunophenotype with molecular confirmation; in particular, NR4A3 break-apart fluorescence in situ hybridisation (FISH) provides a practical single-assay solution. For localised disease, complete surgical excision remains the cornerstone of treatment. Radiotherapy (RT) improves local control when margins are close or tumours are large. Recurrence-free survival (RFS) varies: local recurrence (LR) rates range from 13 to 42% across studies, and distant metastases develop in around 35-45% of patients, primarily in the lungs. The median time to metastasis is approximately 28 months. Overall survival (OS) reflects the typically indolent yet metastatic course: 5-year OS 66-88%, and 10-year disease-specific survival approximately 85%. In advanced disease, anthracycline-based chemotherapy yields a low objective response rate (ORR), although occasional partial responses occur. By contrast, the anti-angiogenic tyrosine kinase inhibitor pazopanib produced an ORR of 18% and a median progression-free survival (PFS) of 19 months in a multicentre phase 2 study (NCT02066285). No clinically validated agents directly target NR4A3. This review summarises contemporary diagnostics and treatment, emphasising high-quality surgery, selective RT, and consideration of anti-angiogenic tyrosine kinase inhibitors in advanced disease.
期刊介绍:
The "Journal of Cancer Research and Clinical Oncology" publishes significant and up-to-date articles within the fields of experimental and clinical oncology. The journal, which is chiefly devoted to Original papers, also includes Reviews as well as Editorials and Guest editorials on current, controversial topics. The section Letters to the editors provides a forum for a rapid exchange of comments and information concerning previously published papers and topics of current interest. Meeting reports provide current information on the latest results presented at important congresses.
The following fields are covered: carcinogenesis - etiology, mechanisms; molecular biology; recent developments in tumor therapy; general diagnosis; laboratory diagnosis; diagnostic and experimental pathology; oncologic surgery; and epidemiology.