Descriptive Epidemiology and Survival Rate of Osteosarcoma: The First National Population-Based Study in the Middle East (2008-2014)

Document Type : RESEARCH PAPER

Authors

1 1 Department of orthopedics, Taleghani Hospital Research Development committee, Shahid Beheshti University of medical sciences, Tehran, Iran 2 Department of Orthopaedic Surgery, University of Pittsburgh, Pittsburgh, PA, USA

2 Department of Orthopedic Surgery, Clinical Research Development Unit of Shohada-e Tajrish Hospital, Shahid Beheshti University of Medical Sciences, Tehran, Iran

3 Bone and Joint reconstruction research center, Shafa Orthopedic Hospital, Iran University of Medical Sciences, Tehran, Iran

4 Cancer Research Center, Shahid Beheshti University of Medical Sciences, Tehran, Iran

Abstract

Objectives: The epidemiology of osteosarcoma (OS), the most common primary bone sarcoma, was not 
evaluated in the Middle East. Therefore, this study aimed to examine the incidence, demographic 
characteristics, epidemiology, and survival rate of patients with different subtypes of OS, based on data 
derived from the Iran National Cancer Registry (INCR) to evaluate the influence of ethnicity and race.
Methods: All OS patients registered in the INCR between March 20, 2008, and March 20, 2014, were enrolled in 
this study, and information such as age, gender, cancer location, OS subtype, and survival time were evaluated 
statistically.
Results: The Age-Standardized Incidence Rate (ASIR) for OS was 3.02 per million person-years, with a mean age 
of 25.6 years and a male-female ratio of 1.54:1. Not Otherwise Specified (NOS) OS, chondroblastic OS, and central 
OS had the highest frequencies among the subtypes of OS. The overall one-, three-, and five-year survival rates 
were 87%, 61%, and 49%, respectively, with a mean duration of 6.16 years.
Conclusion: The ASIR of OS in our country was similar to that in the US and higher than that in China. The peak 
frequency was between 15-19 years old. The male-female ratio in our patients was higher than the OS gender ratio 
in most series. Although it was not statistically significant, older age at the time of diagnosis, axial location, and male 
gender were the poorest prognosis factors.
 Level of evidence: III

Keywords

Main Subjects


  1. Ottaviani G, Jaffe N. The epidemiology of osteosarcoma. Cancer Treat Res. 2009; 152:3-13. doi: 10.1007/978-1-4419-0284-9_1.
  2. Dorfman HD, Czerniak B. Bone cancers. Cancer. 1995; 75(1 Suppl):203-210. doi: 10.1002/1097-0142(19950101)75:1+<203::aid-cncr2820751308>3.0.co;2-v.
  3. Mirabello L, Troisi RJ, Savage SA. Osteosarcoma incidence and survival rates from 1973 to 2004: data from the Surveillance, Epidemiology, and End Results Program. Cancer. 2009; 115(7):1531-1543. doi:10.1002/cncr.24121.
  4. Campanacci M. Bone and soft tissue tumors: clinical features, imaging, pathology and treatment. Springer Science & Business Media; 2013.
  5. Damron TA, Ward WG, Stewart A. Osteosarcoma, chondrosarcoma, and Ewing's sarcoma: National Cancer Data Base Report. Clin Orthop Relat Res. 2007; 459:40-47. doi:10.1097/BLO.0b013e318059b8c9.
  6. Anfinsen KP, Devesa SS, Bray F, et al. Age-period-cohort analysis of primary bone cancer incidence rates in the United States (1976-2005). Cancer Epidemiol Biomarkers Prev. 2011; 20(8):1770-1777. doi:10.1158/1055-9965.EPI-11-0136.
  7. Hayden JB, Hoang BH. Osteosarcoma: basic science and clinical implications. Orthop Clin North Am. 2006;37(1):1-7. doi:10.1016/j.ocl.2005.06.004.
  8. Pingping B, Yuhong Z, Weiqi L, et al. Incidence and Mortality of Sarcomas in Shanghai, China, During 2002-2014. Front Oncol. 2019;9:662.doi:10.3389/fonc.2019.00662.
  9. 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):593-599. doi:10.1016/j.canep.2015.05.001.
  10. Mialou V, Philip T, Kalifa C, et al. Metastatic osteosarcoma at diagnosis: prognostic factors and long-term outcome--the French pediatric experience. Cancer. 2005; 104(5):1100-1109. doi:10.1002/cncr.21263.
  11. Akbari A, Khayamzadeh M, Salmanian R, et al. International Journal of Cancer Management. National cancer mortality-to-incidence ratio (MIR) in Iran (2005-2014). 2019; 12(6). doi.org/10.5812/ijcm.94145.
  12. Trott PA. International classification of diseases for oncology. Journal of clinical pathology. 1977;30(8):782.
  13. Ahmad OB, Boschi-Pinto C, Lopez AD, Murray CJ, Lozano R, Inoue M. Age standardization of rates: a new WHO standard. Geneva: World Health Organization. 2001; 9(10):1-4.
  14. Boyle P, Parkin DM. Cancer registration: principles and methods. Statistical methods for registries. IARC Sci Publ. 1991 ;( 95):126-158.
  15. Duong LM, Richardson LC. Descriptive epidemiology of malignant primary osteosarcoma using population-based registries, United States, 1999-2008. J Registry Manag. 2013; 40(2):59-64.
  16. Whelan J, McTiernan A, Cooper N, et al. Incidence and survival of malignant bone sarcomas in England 1979-2007. Int J Cancer. 2012; 131(4):E508-E517. doi:10.1002/ijc.26426.
  17. Jaffe N, Bch MB, Paed D. Malignant bone tumors in children: incidence and etiologic considerations. Solid Tumors in Childhood. Littleton, MA: PSG Publishing Co. 1979:1-0.
  18. Mirabello L, Troisi RJ, Savage SA. International osteosarcoma incidence patterns in children and adolescents, middle ages and elderly persons. Int J Cancer. 2009; 125(1):229-234. doi:10.1002/ijc.24320.
  19. Hartford CM, Wodowski KS, Rao BN, Khoury JD, Neel MD, Daw NC. Osteosarcoma among children aged 5 years or younger: the St. Jude Children's Research Hospital experience. J Pediatr Hematol Oncol. 2006; 28(1):43-47.
  20. Huvos AG. Osteogenic sarcoma of bones and soft tissues in older persons. A clinicopathologic analysis of 117 patients older than 60 years. Cancer. 1986; 57(7):1442-1449. doi: 10.1002/1097-0142(19860401)57:7<1442::aid-cncr2820570734>3.0.co;2-3.
  21. Hansen MF, Seton M, Merchant A. Osteosarcoma in Paget's disease of bone. J Bone Miner Res. 2006; 21 Suppl 2:P58-P63. doi:10.1359/jbmr.06s211.
  22. Gurney JG, Swensen AR, Bulterys M. Malignant bone tumors. Cancer incidence and survival among children and adolescents: United States SEER Program. 1975; 1995(1999):99-110.
  23. Bleyer AO, O'leary M, Barr R, Ries LA. Cancer epidemiology in older adolescents and young adults 15 to 29 years of age, including SEER incidence and survival: 1975-2000. Cancer epidemiology in older adolescents and young adults 15 to 29 years of age, including SEER incidence and survival: 1975-2000. 2006.
  24. Dahlin DC, Unni KK. Bone tumors: general aspects and data on 8,547 cases. 1986.

25. Larsson SE, Lorentzon R. The incidence of malignant primary bone tumours in relation to age, sex and site. A study of 

  1. Ottaviani G, Jaffe N. The epidemiology of osteosarcoma. Cancer Treat Res. 2009; 152:3-13. doi: 10.1007/978-1-4419-0284-9_1.
  2. Dorfman HD, Czerniak B. Bone cancers. Cancer. 1995; 75(1 Suppl):203-210. doi: 10.1002/1097-0142(19950101)75:1+<203::aid-cncr2820751308>3.0.co;2-v.
  3. Mirabello L, Troisi RJ, Savage SA. Osteosarcoma incidence and survival rates from 1973 to 2004: data from the Surveillance, Epidemiology, and End Results Program. Cancer. 2009; 115(7):1531-1543. doi:10.1002/cncr.24121.
  4. Campanacci M. Bone and soft tissue tumors: clinical features, imaging, pathology and treatment. Springer Science & Business Media; 2013.
  5. Damron TA, Ward WG, Stewart A. Osteosarcoma, chondrosarcoma, and Ewing's sarcoma: National Cancer Data Base Report. Clin Orthop Relat Res. 2007; 459:40-47. doi:10.1097/BLO.0b013e318059b8c9.
  6. Anfinsen KP, Devesa SS, Bray F, et al. Age-period-cohort analysis of primary bone cancer incidence rates in the United States (1976-2005). Cancer Epidemiol Biomarkers Prev. 2011; 20(8):1770-1777. doi:10.1158/1055-9965.EPI-11-0136.
  7. Hayden JB, Hoang BH. Osteosarcoma: basic science and clinical implications. Orthop Clin North Am. 2006;37(1):1-7. doi:10.1016/j.ocl.2005.06.004.
  8. Pingping B, Yuhong Z, Weiqi L, et al. Incidence and Mortality of Sarcomas in Shanghai, China, During 2002-2014. Front Oncol. 2019;9:662.doi:10.3389/fonc.2019.00662.
  9. 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):593-599. doi:10.1016/j.canep.2015.05.001.
  10. Mialou V, Philip T, Kalifa C, et al. Metastatic osteosarcoma at diagnosis: prognostic factors and long-term outcome--the French pediatric experience. Cancer. 2005; 104(5):1100-1109. doi:10.1002/cncr.21263.
  11. Akbari A, Khayamzadeh M, Salmanian R, et al. International Journal of Cancer Management. National cancer mortality-to-incidence ratio (MIR) in Iran (2005-2014). 2019; 12(6). doi.org/10.5812/ijcm.94145.
  12. Trott PA. International classification of diseases for oncology. Journal of clinical pathology. 1977;30(8):782.
  13. Ahmad OB, Boschi-Pinto C, Lopez AD, Murray CJ, Lozano R, Inoue M. Age standardization of rates: a new WHO standard. Geneva: World Health Organization. 2001; 9(10):1-4.
  14. Boyle P, Parkin DM. Cancer registration: principles and methods. Statistical methods for registries. IARC Sci Publ. 1991 ;( 95):126-158.
  15. Duong LM, Richardson LC. Descriptive epidemiology of malignant primary osteosarcoma using population-based registries, United States, 1999-2008. J Registry Manag. 2013; 40(2):59-64.
  16. Whelan J, McTiernan A, Cooper N, et al. Incidence and survival of malignant bone sarcomas in England 1979-2007. Int J Cancer. 2012; 131(4):E508-E517. doi:10.1002/ijc.26426.
  17. Jaffe N, Bch MB, Paed D. Malignant bone tumors in children: incidence and etiologic considerations. Solid Tumors in Childhood. Littleton, MA: PSG Publishing Co. 1979:1-0.
  18. Mirabello L, Troisi RJ, Savage SA. International osteosarcoma incidence patterns in children and adolescents, middle ages and elderly persons. Int J Cancer. 2009; 125(1):229-234. doi:10.1002/ijc.24320.
  19. Hartford CM, Wodowski KS, Rao BN, Khoury JD, Neel MD, Daw NC. Osteosarcoma among children aged 5 years or younger: the St. Jude Children's Research Hospital experience. J Pediatr Hematol Oncol. 2006; 28(1):43-47.
  20. Huvos AG. Osteogenic sarcoma of bones and soft tissues in older persons. A clinicopathologic analysis of 117 patients older than 60 years. Cancer. 1986; 57(7):1442-1449. doi: 10.1002/1097-0142(19860401)57:7<1442::aid-cncr2820570734>3.0.co;2-3.
  21. Hansen MF, Seton M, Merchant A. Osteosarcoma in Paget's disease of bone. J Bone Miner Res. 2006; 21 Suppl 2:P58-P63. doi:10.1359/jbmr.06s211.
  22. Gurney JG, Swensen AR, Bulterys M. Malignant bone tumors. Cancer incidence and survival among children and adolescents: United States SEER Program. 1975; 1995(1999):99-110.
  23. Bleyer AO, O'leary M, Barr R, Ries LA. Cancer epidemiology in older adolescents and young adults 15 to 29 years of age, including SEER incidence and survival: 1975-2000. Cancer epidemiology in older adolescents and young adults 15 to 29 years of age, including SEER incidence and survival: 1975-2000. 2006.
  24. Dahlin DC, Unni KK. Bone tumors: general aspects and data on 8,547 cases. 1986.
  25. Larsson SE, Lorentzon R. The incidence of malignant primary bone tumours in relation to age, sex and site. A study of osteogenic sarcoma, chondrosarcoma and Ewing's sarcoma diagnosed in Sweden from 1958 to 1968. J Bone Joint Surg Br. 1974; 56B (3):534-540.
  26. Gurney JG, Severson RK, Davis S, Robison LL. Incidence of cancer in children in the United States. Sex-, race-, and 1-year age-specific rates by histologic type. Cancer. 1995; 75(8):2186-2195. doi: 10.1002/1097-0142(19950415)75:8<2186::aid-cncr2820750825>3.0.co;2-f.
  27. Mckenna RJ, Schwinn CP, Soong K, et al. Sarcomata of the osteogenic series (osteosarcoma, fibrosarcoma, chondrosarcoma, parosteal osteogenic sarcoma, and sarcomata arising in abnormal bone): an analysis of 552 cases. JBJS. 1966; 48(1):1-26.
  28. Dahlin DC, Coventry MB. Osteogenic sarcoma. A study of six hundred cases. J Bone Joint Surg Am. 1967; 49(1):101-110.
  29. Bacci G, Ferrari S, Bertoni F, et al. Long-term outcome for patients with nonmetastatic osteosarcoma of the extremity treated at the istituto ortopedico rizzoli according to the istituto ortopedico rizzoli/osteosarcoma-2 protocol: an updated report. J Clin Oncol. 2000; 18(24):4016-4027. doi:10.1200/JCO.2000.18.24.4016.
  30. Mankin HJ, Hornicek FJ, Rosenberg AE, Harmon DC, Gebhardt MC. Survival data for 648 patients with osteosarcoma treated at one institution. Clin Orthop Relat Res. 2004 ;( 429):286-291. doi:10.1097/01.blo.0000145991.65770.e6.
  31. Bielack SS, Kempf-Bielack B, Delling G, 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):776-790. doi:10.1200/JCO.2002.20.3.776.
  32. Jawad MU, Cheung MC, Clarke J, Koniaris LG, Scully SP. Osteosarcoma: improvement in survival limited to high-grade patients only. J Cancer Res Clin Oncol. 2011; 137(4):597-607. doi: 10.1007/s00432-010-0923-7.
  33. Tempelaere C, Biau D, Babinet A, Anract P. Osteosarcoma after the age of fifty: A clinicopathological study. Eur J Surg Oncol. 2019; 45(7):1288-1292. doi:10.1016/j.ejso.2019.04.010.
  34. Saeter G, Elomaa I, Wahlqvist Y, et al. Prognostic factors in bone sarcomas. Acta Orthop Scand Suppl. 1997; 273:156-160. doi:10.1080/17453674.1997.11744723.
  35. Burt M. Primary malignant tumors of the chest wall. The Memorial Sloan-Kettering Cancer Center experience. Chest Surg Clin N Am. 1994; 4(1):137-154.
  36. Smeland S, Bielack SS, Whelan J, et al. Survival and prognosis with osteosarcoma: outcomes in more than 2000 patients in the EURAMOS-1 (European and American Osteosarcoma Study) cohort. Eur J Cancer. 2019; 109:36-50. doi:10.1016/j.ejca.2018.11.027.