Abstract
Background: With improved survival, more bone sarcoma survivors are approaching middle age making it crucial to investigate the late effects of their cancer and its treatment. We investigated the long-term risks of adverse outcomes among 5-year bone sarcoma survivors within the British Childhood Cancer Survivor Study. Methods: Cause-specific mortality and risk of subsequent primary neoplasms (SPNs) were investigated for 664 bone sarcoma survivors. Use of health services, health and marital status, alcohol and smoking habits, and educational qualifications were investigated for survivors who completed a questionnaire. Results: Survivors were seven times more likely to experience all-cause mortality than expected, and there were substantial differences in risk depending on tumour type. Beyond 25 years follow-up the risk of dying from all-causes was comparable to the general population. This is in contrast to dying before 25 years where the risk was 12.7-fold that expected. Survivors were also four times more likely to develop a SPN than expected, where the excess was restricted to 5-24 years post diagnosis. Increased health-care usage and poor health status were also found. Nonetheless, for some psychosocial outcomes survivors were better off than expected. Conclusions: Up to 25 years after 5-year survival, bone sarcoma survivors are at substantial risk of death and SPNs, but this is greatly reduced thereafter. As 95% of all excess deaths before 25 years follow-up were due to recurrences and SPNs, increased monitoring of survivors could prevent mortality. Furthermore, bone and breast SPNs should be a particular concern. Since there are variations in the magnitude of excess risk depending on the specific adverse outcome under investigation and whether the survivors were initially diagnosed with osteosarcoma or Ewing sarcoma, risks need to be assessed in relation to these factors. These findings should provide useful evidence for risk stratification and updating clinical follow-up guidelines.
| Original language | English |
|---|---|
| Pages (from-to) | 1857-1865 |
| Number of pages | 9 |
| Journal | British Journal of Cancer |
| Volume | 112 |
| Issue number | 12 |
| DOIs | |
| Publication status | Published - 9 Jun 2015 |
Bibliographical note
Funding Information:The British Childhood Cancer Survivor Study (BCCSS) is a national collaborative undertaking guided by a Steering Group that comprises Professor Douglas Easton (chair), Professor Michael Hawkins, Dr Helen Jenkinson, Dr Meriel Jenney, Dr Raoul Reulen, Professor Kathryn Pritchard-Jones, Professor Michael Stevens, Dr Elaine Sugden, Dr Andrew Toogood, and Dr Hamish Wallace. The BCCSS benefits from the contributions of the Officers, Centers, and individual members of the Children’s Cancer and Leukemia Group and the Regional Pediatric Cancer Registries. The BCCSS acknowledges the collaboration of the Office for National Statistics, the General Register Office for Scotland, the Welsh Cancer Intelligence and Surveillance Unit, the National Health Service Information Centre, the regional cancer registries, health authorities, and area health boards for providing general practitioner names and addresses and the general practitioners nationwide who facilitated direct contact with survivors. We are particularly thankful to all survivors who completed a 40-page questionnaire and all General Practitioners who returned consent forms. The BCCSS would not have been possible without the support of our funders: University of Birmingham, Cancer Research UK, the Kay Kendall Leukaemia Fund, and the European Commission to whom we offer our profound thanks. Finally thanks to all BCCSS staff who have given many years of dedicated work to bring the BCCSS to fruition. This work was supported by grant number C386/A10422 from Cancer Research UK; the Kay Kendall Leukaemia Fund; PanCareSurFup, European 7th Frame-work Programme. Raoul C. Reulen is funded by the National Institute for Health Research.
Publisher Copyright:
© 2015 Cancer Research UK. All rights reserved.
Keywords
- bone cancer
- bone sarcoma
- childhood cancer
- Ewing sarcoma
- late effects
- long-term outcomes
- osteosarcoma
- paediatric cancer