Osteosarcoma Relapse After Combined Modality Therapy: An Analysis of Unselected Patients in the Cooperative Osteosarcoma Study Group (COSS)
Abstract
Purpose: To evaluate the impact of patient, tumor, and treatment-related factors on outcome in unselected patients with recurrent osteosarcoma. Patients and Methods: Five hundred seventy-six consecutive patients who had achieved a first complete surgical remission (CR) during combined-modality therapy on neoadjuvant Cooperative Osteosarcoma Study Group (COSS) protocols and then developed recurrent osteosarcoma were analyzed (median time from biopsy to relapse, 1.6 years; range, 0.1 to 14.3 years). There were 501 patients with metastases, 44 with local recurrences, and 31 with both. Metastases involved lungs (469 patients), bones (90 patients), and/or other sites (54 patients). Results: After a median follow-up of 1.2 years for all patients and 4.2 years for survivors, actuarial overall survival (OS) rates at 2, 5, and 10 years were 0.38, 0.23, and 0.18, respectively. Five-year OS was 0.39 for 339 patients with and 0.00 for 229 patients without a second surgical CR (P < .0001). A long time to relapse, a solitary lesion, and, in the case of pulmonary metastases, unilateral disease and the absence of pleural disruption, were of positive prognostic value in uni- and multivariate analyses, as were a second surgical CR and the use of second-line chemotherapy. Radiotherapy was associated with moderately prolonged survival in patients without a second CR. The very limited prognostic differences associated with the use of second-line chemotherapy appeared to be more pronounced with polychemotherapy. Conclusion: Time to relapse and tumor burden correlate with postrelapse outcome in osteosarcoma. Complete surgery is an essential component of curative second-line therapy. Chemotherapy, particularly chemotherapy with more than one agent, may contribute to limited improvements in outcome.
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Systemic therapy in second and later lines seems to produce a limited prolongation in survival.
“Still, systemic therapy in second and later lines seems to produce a limited prolongation in survival [11, 12], and new therapeutic approaches are needed.”
From Pembrolizumab in advanced osteosarcoma: results of a single-arm, open-label, phase 2 trial (2021) · cites this paper as [12]
Patients with unresectable primary or metastatic disease at diagnosis and those with disease relapse have extremely poor outcomes.
“In addition, patients with unresectable primary or metastatic disease at diagnosis and those with disease relapse have extremely poor outcomes 2,126 .”
From Osteosarcoma (2022) · cites this paper as [126]
Second complete remission, which is only achievable through surgery, has a strong association with improved outcomes after relapse in retrospective studies.
“This second complete remission, which is only achievable through surgery, has a strong association with improved outcomes after relapse in retrospective studies 126 .”
From Osteosarcoma (2022) · cites this paper as [126]
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- Authors
- Kempf-Bielack, B., Bielack, S. S., Jürgens, H., Branscheid, D., Berdel, W. E., Exner, G. U., Göbel, U., Helmke, K., Jundt, G., Kabisch, H., Kevric, M., Klingebiel, T., Kotz, R., Maas, R., Schwarz, R., Semik, M., Treuner, J., Zoubek, A., & Winkler, K.
- Affiliations
- From the Universitätsklinikum Münster, Klinik und Poliklinik für Kinder- und Jugendmedizin, Pädiatrische Hämatologie und Onkologie; Universitätsklinikum Münster, Medizinische Klinik und Poliklinik A; Universitätsklinikum Münster, Klinik und Poliklinik für Thorax-, Herz- und Gefäβchirurgie, Münster; Krankenhaus Groβhansdorf, Zentrum für Pneumologie und Thoraxchirurgie, Groβhansdorf; Universitätsklinikum Düsseldorf, Klinik für Kinder-Onkologie, -Hämatologie und -Immunologie, Düsseldorf;...
- Düsseldorf University Hospital (DE)
- ORCID
- Venue
- Journal of Clinical Oncology
- Published
- 2005-01-20
- Volume
- 23
- Issue
- 3
- Pages
- 559-568
- ISSN
- 0732-183X (ISSN-L)
- 1527-7755
- OpenAlex
- W2141788705
- Cited by
- 643
- References
- 46
- Citations by year
- 2026: 17 · 2025: 19 · 2024: 24 · 2023: 29 · 2022: 43 · 2021: 54 · 2020: 55 · 2019: 27 · 2018: 32 · 2017: 53 · 2016: 38 · 2015: 36 · 2014: 38 · 2013: 25 · 2012: 24
- FWCI
- 13.19
- Type
- journal article
- Language
- en
- Open access
- closed
- Locations
- Journal of Clinical Oncology (publishedVersion)
- PubMed (publishedVersion)
- Funders
- Merck KGaA
- Topics
- Health Sciences › Medicine › Pulmonary and Respiratory Medicine › Sarcoma Diagnosis and Treatment
- Health Sciences › Medicine › Rheumatology › Bone Tumor Diagnosis and Treatments
- Health Sciences › Medicine › Surgery › Management of metastatic bone disease
- Keywords
- osteosarcoma, relapse, recurrent osteosarcoma, multimodal therapy, lung metastasis, prognostic factors, second-line chemotherapy, radiotherapy, overall survival, tumor burden, time to relapse
- MeSH terms
- Adolescent, Adult, Aged, Antineoplastic Combined Chemotherapy Protocols, Bone Neoplasms, Child, Child, Preschool, Combined Modality Therapy, Female, Humans, Male, Middle Aged, Neoplasm Recurrence, Local, Prognosis, Risk Factors, Osteosarcoma, Time Factors, Cohort Studies, Survival Analysis
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