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Open Access 01-12-2018 | Research article

Descriptive epidemiology and outcomes of bone sarcomas in adolescent and young adult patients in Japan

Authors: Takashi Fukushima, Koichi Ogura, Toru Akiyama, Katsushi Takeshita, Akira Kawai

Published in: BMC Musculoskeletal Disorders | Issue 1/2018

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Abstract

Background

There have been fewer improvements in the clinical outcomes of adolescent and young adult (AYA) patients with cancer than for children and older adults, possibly because fewer studies focus on patients in this age group. The aims of this study were (1) to determine survival rates of bone sarcoma among AYAs in Japan (for comparison with other age groups), and (2) to establish whether belonging to the AYA age group at diagnosis was correlated with poor cancer survival in Japan.

Methods

A total of 3457 patients diagnosed with bone sarcoma (1930 male and 1527 female) were identified from 63,931 records in the Bone and Soft Tissue Tumor (BSTT) registry, a nationwide Japanese database, from 2006 to 2013. The histologic subtypes of bone sarcoma were osteosarcoma, chondrosarcoma, and Ewing sarcoma. The primary endpoints for prognosis were the occurrence of tumor-related death. We compared the epidemiological features of AYAs with other age groups. The cancer survival rates were calculated using the Kaplan-Meier method. Cox proportional hazards models were used to analyze the prognostic factors for cancer survival.

Results

The majority of AYA had osteosarcoma 631 (56.2%), while 198 (17.6%) had chondrosarcoma. The frequency of bone sarcoma occurrence was highest among AYA patients, who accounted for a marked proportion of patients with each type of sarcoma. With the exception of sarcoma type, AYA patients did not significantly differ from patients in other age groups for any of the investigated clinicopathological parameters. Cancer survival of AYA patients was significantly higher than in the elderly. Univariate and multivariate analyses revealed that AYA status was not a predictor of poor cancer survival. However, older age (≥65 years) was a predictor of poor cancer survival in patients with overall bone sarcoma, osteosarcoma, chondrosarcoma.

Conclusion

This epidemiological study is the first to investigate AYA patients with bone sarcoma using the nationwide BSTT Registry. We found that cancer survival of AYA patients was significantly higher than that of the elderly. AYA status was not a predictor of poor cancer survival in Japan.
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Literature
1.
2.
go back to reference Albritton K, Bleyer WA. The management of cancer in the older adolescent. Eur J Cancer. 2003;39(18):2584–99.CrossRefPubMed Albritton K, Bleyer WA. The management of cancer in the older adolescent. Eur J Cancer. 2003;39(18):2584–99.CrossRefPubMed
3.
go back to reference Bleyer A, Budd T, Montello M. Adolescents and young adults with cancer: the scope of the problem and criticality of clinical trials. Cancer. 2006;107(7 Suppl):1645–55.CrossRefPubMed Bleyer A, Budd T, Montello M. Adolescents and young adults with cancer: the scope of the problem and criticality of clinical trials. Cancer. 2006;107(7 Suppl):1645–55.CrossRefPubMed
4.
go back to reference Thomas DM, Albritton KH, Ferrari A. Adolescent and young adult oncology: an emerging field. J Clin Oncol. 2010;28(32):4781–2.CrossRefPubMed Thomas DM, Albritton KH, Ferrari A. Adolescent and young adult oncology: an emerging field. J Clin Oncol. 2010;28(32):4781–2.CrossRefPubMed
5.
go back to reference Children’s Oncology Group, SEER Program (National Cancer Institute (U.S.)). Cancer epidemiology in older adolescents and young adults 15 to 29 years of age : including SEER incidence and survival, 1975–2000. Bethesda: U.S. Dept. of Health and Human Services, National Institutes of Health, National Cancer Institute; 2006. x, 205 p. p. Children’s Oncology Group, SEER Program (National Cancer Institute (U.S.)). Cancer epidemiology in older adolescents and young adults 15 to 29 years of age : including SEER incidence and survival, 1975–2000. Bethesda: U.S. Dept. of Health and Human Services, National Institutes of Health, National Cancer Institute; 2006. x, 205 p. p.
6.
go back to reference Stiller CA, Trama A, Serraino D, Rossi S, Navarro C, Chirlaque MD, et al. Descriptive epidemiology of sarcomas in Europe: report from the RARECARE project. Eur J Cancer. 2013;49(3):684–95.CrossRefPubMed Stiller CA, Trama A, Serraino D, Rossi S, Navarro C, Chirlaque MD, et al. Descriptive epidemiology of sarcomas in Europe: report from the RARECARE project. Eur J Cancer. 2013;49(3):684–95.CrossRefPubMed
7.
go back to reference Eleuterio SJ, Senerchia AA, Almeida MT, Da Costa CM, Lustosa D, Calheiros LM, et al. Osteosarcoma in patients younger than 12 years old without metastases have similar prognosis as adolescent and young adults. Pediatr Blood Cancer. 2015;62(7):1209–13.CrossRefPubMed Eleuterio SJ, Senerchia AA, Almeida MT, Da Costa CM, Lustosa D, Calheiros LM, et al. Osteosarcoma in patients younger than 12 years old without metastases have similar prognosis as adolescent and young adults. Pediatr Blood Cancer. 2015;62(7):1209–13.CrossRefPubMed
8.
go back to reference Haggar FA, Preen DB, Pereira G, Holman CD, Einarsdottir K. Cancer incidence and mortality trends in Australian adolescents and young adults, 1982-2007. BMC Cancer. 2012;12:151.CrossRefPubMedPubMedCentral Haggar FA, Preen DB, Pereira G, Holman CD, Einarsdottir K. Cancer incidence and mortality trends in Australian adolescents and young adults, 1982-2007. BMC Cancer. 2012;12:151.CrossRefPubMedPubMedCentral
9.
go back to reference Herzog CE. Overview of sarcomas in the adolescent and young adult population. J Pediatr Hematol Oncol. 2005;27(4):215–8.CrossRefPubMed Herzog CE. Overview of sarcomas in the adolescent and young adult population. J Pediatr Hematol Oncol. 2005;27(4):215–8.CrossRefPubMed
10.
go back to reference Keegan TH, Ries LA, Barr RD, Geiger AM, Dahlke DV, Pollock BH, et al. Comparison of cancer survival trends in the United States of adolescents and young adults with those in children and older adults. Cancer. 2016;122(7):1009–16.CrossRefPubMed Keegan TH, Ries LA, Barr RD, Geiger AM, Dahlke DV, Pollock BH, et al. Comparison of cancer survival trends in the United States of adolescents and young adults with those in children and older adults. Cancer. 2016;122(7):1009–16.CrossRefPubMed
11.
go back to reference Khamly KK, Thursfield VJ, Fay M, Desai J, Toner GC, Choong PF, et al. Gender-specific activity of chemotherapy correlates with outcomes in chemosensitive cancers of young adulthood. Int J Cancer. 2009;125(2):426–31.CrossRefPubMed Khamly KK, Thursfield VJ, Fay M, Desai J, Toner GC, Choong PF, et al. Gender-specific activity of chemotherapy correlates with outcomes in chemosensitive cancers of young adulthood. Int J Cancer. 2009;125(2):426–31.CrossRefPubMed
12.
go back to reference Ogura K, Higashi T, Kawai A. Statistics of bone sarcoma in Japan: report from the bone and soft tissue tumor registry in Japan. J Orthop Sci. 2017;22(1):133–43.CrossRefPubMed Ogura K, Higashi T, Kawai A. Statistics of bone sarcoma in Japan: report from the bone and soft tissue tumor registry in Japan. J Orthop Sci. 2017;22(1):133–43.CrossRefPubMed
14.
go back to reference Grier HE, Krailo MD, Tarbell NJ, Link MP, Fryer CJ, Pritchard DJ, et al. Addition of ifosfamide and etoposide to standard chemotherapy for Ewing's sarcoma and primitive neuroectodermal tumor of bone. N Engl J Med. 2003;348(8):694–701.CrossRefPubMed Grier HE, Krailo MD, Tarbell NJ, Link MP, Fryer CJ, Pritchard DJ, et al. Addition of ifosfamide and etoposide to standard chemotherapy for Ewing's sarcoma and primitive neuroectodermal tumor of bone. N Engl J Med. 2003;348(8):694–701.CrossRefPubMed
15.
go back to reference Bielack SS, Kempf-Bielack B, Delling G, Exner GU, Flege S, Helmke K, et al. Prognostic factors in high-grade osteosarcoma of the extremities or trunk: an analysis of 1,702 patients treated on neoadjuvant cooperative osteosarcoma study group protocols. J Clin Oncol. 2002;20(3):776–90.CrossRefPubMed Bielack SS, Kempf-Bielack B, Delling G, Exner GU, Flege S, Helmke K, et al. Prognostic factors in high-grade osteosarcoma of the extremities or trunk: an analysis of 1,702 patients treated on neoadjuvant cooperative osteosarcoma study group protocols. J Clin Oncol. 2002;20(3):776–90.CrossRefPubMed
16.
go back to reference Giuffrida AY, Burgueno JE, Koniaris LG, Gutierrez JC, Duncan R, Scully SP. Chondrosarcoma in the United States (1973 to 2003): an analysis of 2890 cases from the SEER database. J Bone Joint Surg Am. 2009;91(5):1063–72.CrossRefPubMed Giuffrida AY, Burgueno JE, Koniaris LG, Gutierrez JC, Duncan R, Scully SP. Chondrosarcoma in the United States (1973 to 2003): an analysis of 2890 cases from the SEER database. J Bone Joint Surg Am. 2009;91(5):1063–72.CrossRefPubMed
17.
go back to reference Tricoli JV, Seibel NL, Blair DG, Albritton K, Hayes-Lattin B. Unique characteristics of adolescent and young adult acute lymphoblastic leukemia, breast cancer, and colon cancer. J Natl Cancer Inst. 2011;103(8):628–35.CrossRefPubMedPubMedCentral Tricoli JV, Seibel NL, Blair DG, Albritton K, Hayes-Lattin B. Unique characteristics of adolescent and young adult acute lymphoblastic leukemia, breast cancer, and colon cancer. J Natl Cancer Inst. 2011;103(8):628–35.CrossRefPubMedPubMedCentral
18.
go back to reference Pfreundschuh M, Trumper L, Osterborg A, Pettengell R, Trneny M, Imrie K, et al. CHOP-like chemotherapy plus rituximab versus CHOP-like chemotherapy alone in young patients with good-prognosis diffuse large-B-cell lymphoma: a randomised controlled trial by the MabThera international trial (MInT) group. Lancet Oncol. 2006;7(5):379–91.CrossRefPubMed Pfreundschuh M, Trumper L, Osterborg A, Pettengell R, Trneny M, Imrie K, et al. CHOP-like chemotherapy plus rituximab versus CHOP-like chemotherapy alone in young patients with good-prognosis diffuse large-B-cell lymphoma: a randomised controlled trial by the MabThera international trial (MInT) group. Lancet Oncol. 2006;7(5):379–91.CrossRefPubMed
19.
go back to reference Roy L, Guilhot J, Krahnke T, Guerci-Bresler A, Druker BJ, Larson RA, et al. Survival advantage from imatinib compared with the combination interferon-alpha plus cytarabine in chronic-phase chronic myelogenous leukemia: historical comparison between two phase 3 trials. Blood. 2006;108(5):1478–84.CrossRefPubMed Roy L, Guilhot J, Krahnke T, Guerci-Bresler A, Druker BJ, Larson RA, et al. Survival advantage from imatinib compared with the combination interferon-alpha plus cytarabine in chronic-phase chronic myelogenous leukemia: historical comparison between two phase 3 trials. Blood. 2006;108(5):1478–84.CrossRefPubMed
20.
go back to reference Adams SH, Newacheck PW, Park MJ, Brindis CD, Irwin CE Jr. Health insurance across vulnerable ages: patterns and disparities from adolescence to the early 30s. Pediatrics. 2007;119(5):e1033–9.CrossRefPubMed Adams SH, Newacheck PW, Park MJ, Brindis CD, Irwin CE Jr. Health insurance across vulnerable ages: patterns and disparities from adolescence to the early 30s. Pediatrics. 2007;119(5):e1033–9.CrossRefPubMed
21.
go back to reference Keegan TH, Tao L, DeRouen MC, Wu XC, Prasad P, Lynch CF, et al. Medical care in adolescents and young adult cancer survivors: what are the biggest access-related barriers? J Cancer Surviv. 2014;8(2):282–92.CrossRefPubMedPubMedCentral Keegan TH, Tao L, DeRouen MC, Wu XC, Prasad P, Lynch CF, et al. Medical care in adolescents and young adult cancer survivors: what are the biggest access-related barriers? J Cancer Surviv. 2014;8(2):282–92.CrossRefPubMedPubMedCentral
22.
go back to reference Akiyama T, Saita K, Chikuda H, Horiguchi H, Fushimi K, Yasunaga H. Mortality and morbidity following surgery for primary malignant musculoskeletal tumors in the pelvis and limbs: a retrospective analysis using the Japanese diagnosis procedure combination database. J Cancer Ther. 2016;07(04):303–10.CrossRef Akiyama T, Saita K, Chikuda H, Horiguchi H, Fushimi K, Yasunaga H. Mortality and morbidity following surgery for primary malignant musculoskeletal tumors in the pelvis and limbs: a retrospective analysis using the Japanese diagnosis procedure combination database. J Cancer Ther. 2016;07(04):303–10.CrossRef
23.
go back to reference Ogura K, Yasunaga H, Horiguchi H, Ohe K, Shinoda Y, Tanaka S, et al. Impact of hospital volume on postoperative complications and in-hospital mortality after musculoskeletal tumor surgery: analysis of a national administrative database. J Bone Joint Surg Am. 2013;95(18):1684–91.CrossRefPubMed Ogura K, Yasunaga H, Horiguchi H, Ohe K, Shinoda Y, Tanaka S, et al. Impact of hospital volume on postoperative complications and in-hospital mortality after musculoskeletal tumor surgery: analysis of a national administrative database. J Bone Joint Surg Am. 2013;95(18):1684–91.CrossRefPubMed
24.
go back to reference Ferrari S, Ruggieri P, Cefalo G, Tamburini A, Capanna R, Fagioli F, et al. Neoadjuvant chemotherapy with methotrexate, cisplatin, and doxorubicin with or without ifosfamide in nonmetastatic osteosarcoma of the extremity: an Italian sarcoma group trial ISG/OS-1. J Clin Oncol. 2012;30(17):2112–8.CrossRefPubMed Ferrari S, Ruggieri P, Cefalo G, Tamburini A, Capanna R, Fagioli F, et al. Neoadjuvant chemotherapy with methotrexate, cisplatin, and doxorubicin with or without ifosfamide in nonmetastatic osteosarcoma of the extremity: an Italian sarcoma group trial ISG/OS-1. J Clin Oncol. 2012;30(17):2112–8.CrossRefPubMed
25.
go back to reference Kudawara I, Aoki Y, Ueda T, Araki N, Naka N, Nakanishi H, et al. Neoadjuvant and adjuvant chemotherapy with high-dose ifosfamide, doxorubicin, cisplatin and high-dose methotrexate in non-metastatic osteosarcoma of the extremities: a phase II trial in Japan. J Chemother. 2013;25(1):41–8.CrossRefPubMed Kudawara I, Aoki Y, Ueda T, Araki N, Naka N, Nakanishi H, et al. Neoadjuvant and adjuvant chemotherapy with high-dose ifosfamide, doxorubicin, cisplatin and high-dose methotrexate in non-metastatic osteosarcoma of the extremities: a phase II trial in Japan. J Chemother. 2013;25(1):41–8.CrossRefPubMed
26.
go back to reference Whelan JS, Bielack SS, Marina N, Smeland S, Jovic G, Hook JM, et al. EURAMOS-1, an international randomised study for osteosarcoma: results from pre-randomisation treatment. Ann Oncol. 2015;26(2):407–14.CrossRefPubMed Whelan JS, Bielack SS, Marina N, Smeland S, Jovic G, Hook JM, et al. EURAMOS-1, an international randomised study for osteosarcoma: results from pre-randomisation treatment. Ann Oncol. 2015;26(2):407–14.CrossRefPubMed
27.
go back to reference Bieling P, Rehan N, Winkler P, Helmke K, Maas R, Fuchs N, et al. Tumor size and prognosis in aggressively treated osteosarcoma. J Clin Oncol. 1996;14(3):848–58.CrossRefPubMed Bieling P, Rehan N, Winkler P, Helmke K, Maas R, Fuchs N, et al. Tumor size and prognosis in aggressively treated osteosarcoma. J Clin Oncol. 1996;14(3):848–58.CrossRefPubMed
28.
go back to reference Duchman KR, Gao Y, Miller BJ. Prognostic factors for survival in patients with Ewing’s sarcoma using the surveillance, epidemiology, and end results (SEER) program database. Cancer Epidemiol. 2015;39(2):189–95.CrossRefPubMed Duchman KR, Gao Y, Miller BJ. Prognostic factors for survival in patients with Ewing’s sarcoma using the surveillance, epidemiology, and end results (SEER) program database. Cancer Epidemiol. 2015;39(2):189–95.CrossRefPubMed
29.
go back to reference Bleyer A, Barr R, Hayes-Lattin B, Thomas D, Ellis C, Anderson B, et al. The distinctive biology of cancer in adolescents and young adults. Nat Rev Cancer. 2008;8(4):288–98.CrossRefPubMed Bleyer A, Barr R, Hayes-Lattin B, Thomas D, Ellis C, Anderson B, et al. The distinctive biology of cancer in adolescents and young adults. Nat Rev Cancer. 2008;8(4):288–98.CrossRefPubMed
30.
31.
go back to reference Bertrand TE, Cruz A, Binitie O, Cheong D, Letson GD. Do surgical margins affect local recurrence and survival in extremity, nonmetastatic, high-grade osteosarcoma? Clin Orthop Relat Res. 2016;474(3):677–83.CrossRefPubMed Bertrand TE, Cruz A, Binitie O, Cheong D, Letson GD. Do surgical margins affect local recurrence and survival in extremity, nonmetastatic, high-grade osteosarcoma? Clin Orthop Relat Res. 2016;474(3):677–83.CrossRefPubMed
32.
go back to reference Buchner M, Bernd L, Zahlten-Hinguranage A, Sabo D. Primary malignant tumours of bone and soft tissue in the elderly. Eur J Surg Oncol. 2004;30(8):877–83.CrossRefPubMed Buchner M, Bernd L, Zahlten-Hinguranage A, Sabo D. Primary malignant tumours of bone and soft tissue in the elderly. Eur J Surg Oncol. 2004;30(8):877–83.CrossRefPubMed
33.
go back to reference Duchman KR, Gao Y, Miller BJ. Prognostic factors for survival in patients with high-grade osteosarcoma using the surveillance, epidemiology, and end results (SEER) program database. Cancer Epidemiol. 2015;39(4):593–9.CrossRefPubMed Duchman KR, Gao Y, Miller BJ. Prognostic factors for survival in patients with high-grade osteosarcoma using the surveillance, epidemiology, and end results (SEER) program database. Cancer Epidemiol. 2015;39(4):593–9.CrossRefPubMed
Metadata
Title
Descriptive epidemiology and outcomes of bone sarcomas in adolescent and young adult patients in Japan
Authors
Takashi Fukushima
Koichi Ogura
Toru Akiyama
Katsushi Takeshita
Akira Kawai
Publication date
01-12-2018
Publisher
BioMed Central
Published in
BMC Musculoskeletal Disorders / Issue 1/2018
Electronic ISSN: 1471-2474
DOI
https://doi.org/10.1186/s12891-018-2217-1