Demographics
| For efficacy analysis (n=41) | P (Cox univariate analysis for PFS) | Excluded from efficacy analysis (n=2) | |
| Patients | 41 (100.0%) | 2 (100.0%) | |
| Age* (years) median (min, max) | 19 (11–43) | 0.39 | (15–62) |
| Child | 5 (12.2%) | 0 (0%) | |
| Adolescent | 16 (39.0%) | 1 (50.0%) | |
| Adult | 20 (48.8%) | 1 (50.0%) | |
| Gender | 0.78 | ||
| Male | 30 (73.2%) | 1 (50.0%) | |
| Female | 11 (26.8%) | 1 (50.0%) | |
| ECOG performance status at enrollment | 0.22 | ||
| 0 | 34 (82.9%) | 0 (0%) | |
| 1 | 7 (17.1%) | 2 (100.0%) | |
| Presence of metastasis | 0.14 | ||
| No (locally advanced disease) | 2 (4.9%) | 0 (0%) | |
| Yes | 39 (95.1%) | 2 (100.0%) | |
| Primary tumor location | 0.08 | ||
| Distal femur | 13 (31.7%) | 1 (50.0%) | |
| Proximal tibia and fibula | 12 (29.3%) | 0 (0%) | |
| Proximal humerus | 8 (19.5%) | 0 (0%) | |
| Proximal femur | 1 (2.4%) | 0 (0%) | |
| Axial skeleton | 4 (9.8%) | 1 (50.0%) | |
| Others† | 3 (7.3%) | 0 (0%) | |
| Sites of target and non-target lesions | 0.001 | ||
| Lung only | 18 (43.9%) | 0 (0%) | |
| Bone only | 3 (7.3%) | 0 (0%) | |
| Lung and bone or viscera | 20 (48.8%) | 2 (100.0%) | |
| Lines of previous chemotherapy including MAP/I‡ | 0.45 | ||
| 1 | 36 (87.8%) | 1 (50.0%) | |
| ≥2 | 5 (12.2%) | 1 (50.0%) | |
| Previous radiotherapy | 0.65 | ||
| No | 39 (95.1%) | 2 (100.0%) | |
| Yes, not the target lesion | 2 (4.9%) | 0 (0%) | |
| High-grade osteosarcoma subtypes | 0.29 | ||
| Common (osteoblastic, chondroblastic, fibroblastic) | 40 (97.6%) | 2 (100.0%) | |
| Small cell | 1 (2.4%) | 0 (0%) | |
| Baseline NLR ≥3 | 41 (100.0%) | N/A | 2 (100.0%) |
| Baseline NLR <3 | 0 (0%) | 0 (0%) | |
| Whether PT declined during treatment | 0.001 | ||
| Yes | 32 (78.0%) | N/A | |
| No | 9 (22.0%) | N/A | |
| Whether APTT declined during treatment | 0.000 | ||
| Yes | 32 (78.0%) | N/A | |
| No | 9 (22.0%) | N/A | |
| Whether PLT elevated during treatment | 0.000 | ||
| Yes | 35 (85.4%) | N/A | |
| No | 6 (14.6%) | N/A | |
| Whether D-dimer elevated during treatment | 0.74 | ||
| Yes | 22 (53.7%) | N/A | |
| No | 18 (43.9%) | N/A | |
| Not available or missing | 1 (2.4%) |
*Groups defined according to Collins et al : child (0–12 for males and 0–11 for females), adolescent (13–17 for males and 12–16 for females), and adult (≥18 for males and ≥17 for females).29
†Others including one polycentric osteosarcoma, one located at foot and one located distal radius.
‡MAP/I, including high-dose methotrexate, doxorubicin, cisplatin with or without ifosfamide. We defined these four agents as first-line chemotherapy.
§Alkaline phosphatase cut-off value according to Bacci et al , defined as: cut-off: 2–10 years 350 IU/L; 10–13 years female 400 IU/L; 13–15 years male 500 IU/L; 20–50 years 100 IU/L; other childhood age 300 IU/L.
AE, adverse events; APTT, activate partial thromboplastin time; ECOG, Eastern Cooperative Oncology Group; N/A, not available; NLR, neutrophil lymphocyte ratio; PFS, progression-free survival; PLT, platelet; PT, prothrombin time; TSH, thyroid-stimulating hormone.