Osteosarcoma Arising as a Secondary Malignancy following Treatment for Hematologic Cancer: A Report of 33 Affected Patients from the Cooperative Osteosarcoma Study Group (COSS)
Abstract
Simple Summary
Abstract
1. Introduction
2. Patients and Methods
2.1. Patient Selection and Data Collection
2.2. Statistical Analyses
3. Results
Patients
- -
- 20 (61%) lymphomas (13 Hodgkin (HL), 7 non-Hodgkin (NHL), 3 diffuse large B-cell (DLBCL), 1 anaplastic large-cell (ALCL), 1 lymphoblastic, 1 mucosa-associated lymphoid tissue (MALT), and 1 unspecified NHL). Among the 20 lymphomas, 16 (80%) were known to have involved the trunk, 4 (20%) the head and neck (incl. 1 with involvement of both regions), and 1 (5%) an extremity.
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- 13 (29%) leukemias (12 acute lymphoblastic (ALL)—7 B-precursor, 4 T-, 1 unspecified; 1 acute myeloid leukemia (AML)).
- -
- 1 benign tumor (adenoma of the thyroid treated by surgery);
- -
- 3 borderline tumors (2 basal cell carcinomas of the skin, 1 phylloides tumors of the breast, all treated by surgery only);
- -
- 1 solid malignancy (1 breast cancer treated by surgery and local radiotherapy);
- -
- 2 hematologic malignancies (1 untreated large-cell NHL, 1 chronic lymphocytic leukemia treated by chemotherapy).
4. Discussion
5. Conclusions
Author Contributions
Funding
Institutional Review Board Statement
Informed Consent Statement
Data Availability Statement
Conflicts of Interest
References
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No. | Gender | Hematological Malignancy | Intercurrent | |||
---|---|---|---|---|---|---|
Age | Type | Radiotherapy | Chemotherapy | Neoplasm | ||
1 | male | 14.2 | HL | yes | none | none |
2 | male | 2.9 | NHL (NFS) | yes | yes | none |
3 | male | 29.7 | HL | yes | none | none |
4 | male | 12.6 | HL | yes | yes | none |
5 | male | 16.6 | HL | yes | yes | none |
6 | male | 10.8 | ALL (B-precursor) | yes (TBI) | yes | none |
7 | female | 20.8 | HL | yes | none | none |
8 | male | 15.1 | HL | yes | yes | none |
9 | male | 5.6 | ALL (B-precursor) | none | yes | none |
10 | male | 7.4 | ALL (NFS) | yes (TBI) | yes | none |
11 1 | male | 9.4 | NHL (ALCL) | none | yes | NHL (B-large-cell) |
12 | male | 5.8 | HL | yes | yes | none |
13 | female | 10.5 | ALL (T-) | yes (TBI) | yes | none |
14 | male | 12.9 | ALL (T-) | yes | yes | none |
15 | male | 33.4 | HL | yes | none | basal-cell carcinoma |
16 | male | 22.4 | HL | yes | yes | none |
17 | female | 49.8 | NHL (DLBCL) | yes | yes | none |
18 | male | 37.1 | HL | yes | none | none |
19 | male | 0.8 | ALL (B-precursor) | yes (TBI) | yes | none |
20 | female | 22.6 | HL | yes | yes | none |
21 | male | 58.2 | NHL (MALT) | yes | none | CLL |
22 | female | 38.1 | NHL (DLBCL) | yes | yes | none |
23 | male | 3.3 | ALL (T-) | yes (TBI) | yes | none |
24 | male | 15.0 | HL | none | yes | none |
25 | male | 1.4 | ALL (B-precursor) | yes (TBI) | yes | thyroid adenoma |
26 | female | 1.2 | ALL (B-precursor) | yes (TBI) | yes | none |
27 | male | 9.4 | ALL (T-) | yes (TBI) | yes | none |
28 | female | 45.7 | NHL (DLBC) | yes | none | breast cancer |
29 | male | 11.1 | NHL (lymphoblastic) | yes | yes | basal-cell carcinoma |
30 2 | female | 5.4 | ALL (B-precursor) | none | yes | none |
31 2 | female | 12.4 | ALL (B-precursor) | yes (TBI) | yes | phylloides tumor |
32 | female | 4.0 | AML | ND | yes | none |
33 | m | 15.3 | HL | yes | yes | none |
No. | Inter- | Osteosarcoma | Follow-up | |||||||||
---|---|---|---|---|---|---|---|---|---|---|---|---|
val | Age | Type | RX-rel | Prim mets | Surg | Response | Rx | Chem | Years | Further neopl | Outcome | |
1 | 5.5 | 19.7 | HGC | yes | none | yes | good | none | yes | 4.1 | none | DOD 2nd rec |
2 | 9.8 | 12.7 | EOS | yes | none | no | NOP | none | yes | 0.8 | none | DOD no CR |
3 | 18.0 | 47.7 | EOS | yes | none | yes | POP | none | yes | 3.1 | none | DOD 1st rec |
4 | 4.6 | 17.2 | HGC | yes | none | yes | good | none | yes | 4.6 | none | alive CR1 |
5 | 19.0 | 35.6 | HGC | yes | none | yes | NFS | none | yes | 3.1 | none | DOD 1st rec |
6 | 8.2 | 19.0 | HGC | yes | none | no | NOP | yes | yes | 7.2 | MDS | DOD SMD 1 |
7 | 17.7 | 38.5 | HGC | yes | none | yes | poor | none | yes | 1.5 | none | DOD 1st rec |
8 | 3.9 | 18.9 | HGC | yes | none | yes | good | none | yes | 11.0 | none | alive CR2 |
9 | 7.5 | 13.1 | HGC | no | lung | yes | good | none | yes | 6.2 | none | DOD 2nd rec |
10 | 8.8 | 16.1 | POS | yes | bone | yes | poor | none | yes | 4.4 | none | DOD 1st rec |
11 | 5.3 | 14.7 | HGC | no | none | yes | good | none | yes | 7.2 | T-ALL | DOC SMD 2 |
12 | 13.3 | 19.1 | HGC | yes | none | yes | poor | none | yes | 8.7 | none | alive CR3 |
13 | 3.2 | 13.6 | HGC | yes | lung | yes | poor | none | yes | 2.4 | none | DOD 1st rec |
14 | 7.0 | 19.8 | HGC | no | lung | yes | NFS | none | yes | 3.5 | none | DOD 2nd rec |
15 | 27.1 | 60.5 | HGC | yes | none | yes | poor | none | yes | 12.0 | SCC | alive CR1 3 |
16 | 16.1 | 38.6 | UPS | no | none | yes | good | none | yes | 8.8 | BCC | alive CR1 3 |
17 | 6.9 | 56.7 | UPS | yes | none | yes | POP | yes | yes | 10.5 | none | alive CR1 |
18 | 24.4 | 61.5 | HGC | yes | lung, bone | yes | POP | none | yes | 0.9 | none | DOD no CR |
19 | 13.7 | 14.5 | HGC | yes | none | yes | good | none | yes | 10.1 | none | alive CR1 |
20 | 7.7 | 30.3 | HGC | yes | none | no | NFS | NFS | NFS | 0.4 | none | alive no CR |
21 | 11.4 | 69.6 | HGC | yes | lung, bone | no | NFS | NFS | NFS | 0.4 | B-NHL 4 | DUC no CR |
22 | 11.2 | 49.3 | HGC | yes | none | yes | POP | none | yes | 1.1 | none | alive CR1 |
23 | 13.9 | 17.2 | HGC | yes | none | yes | poor | none | yes | 2.8 | none | DOD 2nd rec |
24 | 0.5 | 15.5 | HGC | no | lung | yes | good | none | yes | 10.3 | none | alive CR1 |
25 | 16.6 | 18.1 | POS | yes | none | yes | poor | none | yes | 3.9 | none | alive CR1 |
26 | 5.6 | 6.8 | HGC | yes | none | yes | poor | none | yes | 8.5 | none | alive CR1 |
27 | 9.5 | 18.9 | HGC | yes | none | yes | NFS | none | yes | 5.0 | none | alive CR1 |
28 | 19.3 | 65.0 | HGC | yes | none | yes | poor | none | yes | 2.3 | none | alive CR1 |
29 | 41.6 | 52.7 | HGC | yes | lung | yes | NFS | yes | yes | 2.6 | none | alive no CR |
30 | 9.3 | 14.8 | HGC | no | none | yes | NFS | none | yes | 4.9 | none | alive CR1 |
31 | 9.4 | 21.9 | HGC | yes | none | no | NOP | none | yes | 1.2 | none | alive no CR |
32 | 6.0 | 9.9 | HGC | NFS | none | yes | good | none | yes | 3.2 | none | DOD 1st rec |
33 | 7.8 | 23.1 | HGC | yes | none | no | NOP | none | yes | 0.3 | none | alive no CR |
Patients (%) | 5-Year Overall Survival | 5-Year Event-Free Survival | |||
---|---|---|---|---|---|
Estimate (SE) | p 1 | Estimate (SE) | p 1 | ||
all | |||||
all eligible patients | 33 (100%) | 56% (10%) | - | 40% (9%) | - |
gender | |||||
male | 23 (70%) | 57% (11%) | 0.831 | 39% (10%) | 0.71 |
female | 10 (30%) | 51% (20%) | 44% (18%) | ||
age at leukemia/lymphoma | |||||
below 12 years | 15 (45%) | 64% (13%) | 0.991 | 40% (13%) | 0.983 |
12 years and above | 18 (55%) | 48% (14%) | 40% (12%) | ||
type of hematological malignancy | |||||
lymphoma | 20 (61%) | 57% (13%) | 0.856 | 42% (12%) | 0.735 |
leukemia | 13 (39%) | 57% (15%) | 39% (14%) | ||
chemotherapy for hematological malignancy | |||||
yes | 26 (79%) | 67% (10%) | 0.024 | 45% (10%) | 0.477 |
no | 7 (21%) | 19% (17%) | 19% (17%) | ||
interval hematological malignancy to osteosarcoma | |||||
below 10 years | 19 (58%) | 63% (12%) | 0.736 | 42% (11%) | 0.967 |
10 years or more | 14 (42%) | 49% (15%) | 36% (14%) | ||
age at osteosarcoma | |||||
below 18 years | 12 (36%) | 58% (14%) | 0.797 | 50% (14%) | 0.265 |
18 years and above | 21 (64%) | 55% (13%) | 34% (11%) | ||
osteosarcoma in radiation-field 2,3 | |||||
yes | 26 (81%) | 51% (11%) | 0.596 | 35% (10%) | 0.273 |
no | 6 (19%) | 82% (15%) | 67% (19%) | ||
osteosarcoma site | |||||
extremity | 19 (58%) | 56% (11%) | 0.225 | 56% (12%) | 0.011 |
trunk or head and neck | 14 (42%) | 40% (17%) | 18% (11%) | ||
primary osteosarcoma metastases | |||||
absent | 25 (76%) | 65% (11%) | 0.05 | 49% (10%) | 0.063 |
present | 8 (24%) | 31% (18%) | 13% (12%) | ||
macroscopic surgical remission | |||||
achieved | 25 (76%) | 60% (10%) | 0.017 | NA | |
not achieved | 8 (24%) | 51% (20%) | |||
additional malignancies 4 | |||||
no | 28 (85%) | 52% (11%) | 0.807 | 38% (9%) | 0.893 |
yes | 5 (15%) | 80% (18%) | 60% (22%) |
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Bielack, S.S.; Mettmann, V.; Baumhoer, D.; Blattmann, C.; Burkhardt, B.; Deinzer, C.K.W.; Kager, L.; Kevric, M.; Mauz-Körholz, C.; Müller-Abt, P.; et al. Osteosarcoma Arising as a Secondary Malignancy following Treatment for Hematologic Cancer: A Report of 33 Affected Patients from the Cooperative Osteosarcoma Study Group (COSS). Cancers 2024, 16, 1836. https://doi.org/10.3390/cancers16101836
Bielack SS, Mettmann V, Baumhoer D, Blattmann C, Burkhardt B, Deinzer CKW, Kager L, Kevric M, Mauz-Körholz C, Müller-Abt P, et al. Osteosarcoma Arising as a Secondary Malignancy following Treatment for Hematologic Cancer: A Report of 33 Affected Patients from the Cooperative Osteosarcoma Study Group (COSS). Cancers. 2024; 16(10):1836. https://doi.org/10.3390/cancers16101836
Chicago/Turabian StyleBielack, Stefan S., Vanessa Mettmann, Daniel Baumhoer, Claudia Blattmann, Birgit Burkhardt, Christoph K. W. Deinzer, Leo Kager, Matthias Kevric, Christine Mauz-Körholz, Peter Müller-Abt, and et al. 2024. "Osteosarcoma Arising as a Secondary Malignancy following Treatment for Hematologic Cancer: A Report of 33 Affected Patients from the Cooperative Osteosarcoma Study Group (COSS)" Cancers 16, no. 10: 1836. https://doi.org/10.3390/cancers16101836
APA StyleBielack, S. S., Mettmann, V., Baumhoer, D., Blattmann, C., Burkhardt, B., Deinzer, C. K. W., Kager, L., Kevric, M., Mauz-Körholz, C., Müller-Abt, P., Reinhardt, D., Sabo, A.-A., Schrappe, M., Sorg, B., Windhager, R., & Hecker-Nolting, S. (2024). Osteosarcoma Arising as a Secondary Malignancy following Treatment for Hematologic Cancer: A Report of 33 Affected Patients from the Cooperative Osteosarcoma Study Group (COSS). Cancers, 16(10), 1836. https://doi.org/10.3390/cancers16101836