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Long-term adverse outcomes in survivors of childhood bone sarcoma: the British Childhood Cancer Survivor Study.

Fidler MM, Frobisher C, Guha J, Wong K, Kelly J, Winter DL, Sugden E, Duncan R, Whelan J, Reulen RC, Hawkins MM - Br. J. Cancer (2015)

Bottom Line: Increased health-care usage and poor health status were also found.Nonetheless, for some psychosocial outcomes survivors were better off than expected.These findings should provide useful evidence for risk stratification and updating clinical follow-up guidelines.

View Article: PubMed Central - PubMed

Affiliation: Centre for Childhood Cancer Survivor Studies, School of Health and Population Sciences, Public Health Building, University of Birmingham, Birmingham B15 2TT, UK.

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.

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Cumulative mortality of recurrence and second primary neoplasms among childhood bone sarcoma survivors within the British Childhood Cancer Survivor Study (BCCSS) by tumour type.
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fig1: Cumulative mortality of recurrence and second primary neoplasms among childhood bone sarcoma survivors within the British Childhood Cancer Survivor Study (BCCSS) by tumour type.

Mentions: There was a steep increase in mortality during the initial 5 years following 5-year survival where the cumulative mortality reached 10.4% (95% CI: 8.3–13.0) (Supplementary Figure 1). Subsequently, there was a more gradual incline to 20.6% (95% CI: 17.3–24.3) at 35 years post diagnosis. When stratified by tumour type (Figure 1) a significant difference (P=0.004) in cumulative mortality was observed for recurrences, where Ewing sarcoma survivors had nearly double the cumulative mortality at 35 years post diagnosis (osteosarcoma: 8.5% vs Ewing sarcoma: 16.7%). Conversely, the cumulative mortality due to SPNs was twice as high for osteosarcoma compared with Ewing sarcoma survivors at the same point (osteosarcoma: 6.7% vs Ewing sarcoma: 3.2%).


Long-term adverse outcomes in survivors of childhood bone sarcoma: the British Childhood Cancer Survivor Study.

Fidler MM, Frobisher C, Guha J, Wong K, Kelly J, Winter DL, Sugden E, Duncan R, Whelan J, Reulen RC, Hawkins MM - Br. J. Cancer (2015)

Cumulative mortality of recurrence and second primary neoplasms among childhood bone sarcoma survivors within the British Childhood Cancer Survivor Study (BCCSS) by tumour type.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4580396&req=5

fig1: Cumulative mortality of recurrence and second primary neoplasms among childhood bone sarcoma survivors within the British Childhood Cancer Survivor Study (BCCSS) by tumour type.
Mentions: There was a steep increase in mortality during the initial 5 years following 5-year survival where the cumulative mortality reached 10.4% (95% CI: 8.3–13.0) (Supplementary Figure 1). Subsequently, there was a more gradual incline to 20.6% (95% CI: 17.3–24.3) at 35 years post diagnosis. When stratified by tumour type (Figure 1) a significant difference (P=0.004) in cumulative mortality was observed for recurrences, where Ewing sarcoma survivors had nearly double the cumulative mortality at 35 years post diagnosis (osteosarcoma: 8.5% vs Ewing sarcoma: 16.7%). Conversely, the cumulative mortality due to SPNs was twice as high for osteosarcoma compared with Ewing sarcoma survivors at the same point (osteosarcoma: 6.7% vs Ewing sarcoma: 3.2%).

Bottom Line: Increased health-care usage and poor health status were also found.Nonetheless, for some psychosocial outcomes survivors were better off than expected.These findings should provide useful evidence for risk stratification and updating clinical follow-up guidelines.

View Article: PubMed Central - PubMed

Affiliation: Centre for Childhood Cancer Survivor Studies, School of Health and Population Sciences, Public Health Building, University of Birmingham, Birmingham B15 2TT, UK.

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.

Show MeSH
Related in: MedlinePlus