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. 2021 Apr 2;13(7):1683.
doi: 10.3390/cancers13071683.

Successive Osteosarcoma Relapses after the First Line O2006/Sarcome-09 Trial: What Can We Learn for Further Phase-II Trials?

Eric Thebault  1 , Sophie Piperno-Neumann  2 , Diep Tran  3 , Hélène Pacquement  4 , Perrine Marec-Berard  5 , Cyril Lervat  6 , Marie-Pierre Castex  7 , Morgane Cleirec  8 , Emmanuelle Bompas  9 , Jean-Pierre Vannier  10 , Dominique Plantaz  11 , Laure Saumet  12 , Cecile Verite  13 , Olivier Collard  14 , Claire Pluchart  15 , Claire Briandet  16 , Laure Monard  17 , Laurence Brugieres  1 , Marie-Cécile Le Deley  18 , Nathalie Gaspar  1
Affiliations

Successive Osteosarcoma Relapses after the First Line O2006/Sarcome-09 Trial: What Can We Learn for Further Phase-II Trials?

Eric Thebault et al. Cancers (Basel). .

Abstract

The purpose was to describe first and subsequent relapses in patients from the OS2006/Sarcome-09 trial, to help future trial design. We prospectively collected and analysed relapse data of all French patients included in the OS2006/Sarcome-09 trial, who had achieved a first complete remission. 157 patients experienced a first relapse. The median interval from diagnosis to relapse was 1.7 year (range 0.5-7.6). The first relapse was metastatic in 83% of patients, and disease was not measurable according to RECIST 1.1 criteria in 23%. Treatment consisted in systemic therapy (74%) and surgical resection (68%). A quarter of the patients were accrued in a phase-II clinical trial. A second complete remission was obtained for 79 patients. Most of them had undergone surgery (76/79). The 3-year progression-free and overall survival rates were 21% and 37%, respectively. In patients who achieved CR2, the 3y-PFS and OS rates were 39% and 62% respectively. Individual correlation between subsequent PFS durations was poor. For osteosarcoma relapses, we recommend randomised phase-II trials, open to patients from all age categories (children, adolescents, adults), not limited to patients with measurable disease (but stratified according to disease status), with PFS as primary endpoint, response rate and surgical CR as secondary endpoints.

Keywords: RECIST; metastasis; osteosarcoma; phase-II trial; recurrence/relapse.

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Conflict of interest statement

The authors declare no conflict of interest. The funders had no role in the design of the study; in the collection, analyses, or interpretation of data; in the writing of the manuscript, or in the decision to publish the results.

Figures

Figure 1
Figure 1
Participant flow of the current study.
Figure 2
Figure 2
(A): Outcome after first osteosarcoma relapse. Overall (blue) and progression-free survival (red) of the whole cohort of 157 patients. (B). Outcome after first osteosarcoma relapse: Overall (blue) and progression-free survival (red) of the 39 patients with a unique lung lesion at first relapse. (C). Outcome after first osteosarcoma relapse: Progression-free survival of the 39 patients with a unique lung lesion at first relapse, according whether they received (blue) or not (red) a systemic treatment (chemotherapy).
Figure 2
Figure 2
(A): Outcome after first osteosarcoma relapse. Overall (blue) and progression-free survival (red) of the whole cohort of 157 patients. (B). Outcome after first osteosarcoma relapse: Overall (blue) and progression-free survival (red) of the 39 patients with a unique lung lesion at first relapse. (C). Outcome after first osteosarcoma relapse: Progression-free survival of the 39 patients with a unique lung lesion at first relapse, according whether they received (blue) or not (red) a systemic treatment (chemotherapy).
Figure 3
Figure 3
Correlation between successive PFS. (A). Correlation between PFS2 and PFS1. Kendall’s Tau coefficient = 0.163 (95%CI, 0.043–0.28). GMI > 1.3 = 23.2% (se = 3.6%). (B). Correlation between PFS3 and PFS2. Kendall’s Tau coefficient = 0.088 (95%CI, −0.17–0.33). GMI > 1.3 = 28.6% (se = 8.0%). Blue triangle: without a second event—Red dot: with a second event.
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