HHS Public Access Author manuscript Author Manuscript
Cancer. Author manuscript; available in PMC 2016 March 21. Published in final edited form as: Cancer. 2015 December 15; 121(24): 4279–4281. doi:10.1002/cncr.29683.
Specialty Care for Adult Survivors of Childhood Cancer Emily S. Tonorezos, MD, MPH and Kevin C. Oeffinger, MD Department of Medicine, Memorial Sloan Kettering Cancer Center, New York, New York
Author Manuscript
The field of pediatric oncology has made enormous advances over the past decades, as evidenced in countless scholarly publications and the lay press.1-3 Among the primary beneficiaries of these successes are the more than 400,000 adult survivors of childhood cancer currently living in the United States.4 Unfortunately, late effects of childhood cancer therapy are common, serious, and potentially life-threatening.5,6 Recommended screening and surveillance for late effects are carefully outlined in the Children's Oncology Group guidelines,7,8 but the pool of available oncologists is shrinking,9 and primary care providers express uncertainty regarding how best to care for survivors.10-12 Furthermore, a fragmented approach to health care delivery in the United States results in a haphazard and nonsystematic approach to care for this high-risk population.
Author Manuscript Author Manuscript
In this issue of Cancer, Sutradhar et al13 from Canada have taken advantage of the visionary 1999 decision by the Ontario Ministry of Health and Long-Term Care to initiate a provincial network of specialty clinics for adult survivors of childhood cancer. Using multiple linked administrative health databases, the authors created a population-based cohort of 3912 adult survivors of childhood cancer diagnosed between 1986 and 2005 in Ontario, Canada. Approximately 43% of this cohort attended at least 1 survivor clinic visit during the study period, and 14,187 emergency department (ED) visits were captured. An Andersen-Gill recurrent event multivariate regression model was used, and the results demonstrated that 1 or more prior visits to a survivor clinic was associated with a 19% lower rate of ED visits in comparison with survivors who had not visited a specialty survivor clinic. When a visit to a survivor clinic was considered a cumulative count covariate, each additional visit to a survivor clinic decreased the subsequent ED visit rate by 5%. In further analysis, these results were independent of whether or not the survivor was also seen by a primary care physician, and importantly, both low-acuity and high-acuity ED visits were reduced. On the basis of the strength of these findings, it is reasonable to state that attendance at a specialized survivor clinic results in fewer ED visits; whether underlying morbidity is also affected is an intriguing possibility. The policy, economic, and clinical care implications of these findings are significant. The Ontario Ministry of Health and Long-Term Care made the prescient decision to support the creation of 5 specialty clinics for adult survivors of childhood cancer in 1999. In the United States, a small number of cancer center–based programs for adult survivors of childhood
Corresponding author: Kevin C. Oeffinger, MD, Adult Long-Term Follow-Up Program, Memorial Sloan Kettering Cancer Center, 485 Lexington Avenue, 2nd Floor, New York, NY 10017; Fax: (214) 648-2551; [email protected]. Conflict of Interest Disclosures: The authors made no disclosure.
Tonorezos and Oeffinger
Page 2
Author Manuscript
cancer exist, but their reach is limited, and reimbursement for services can be difficult.14 Reducing ED visits for survivors could be achieved with an expansion of specialty clinics in the United States, perhaps via a Medicare approval process, as is already in place for intensive cardiac rehabilitation programs and other facities.15 though it could be less problematical, establishing survivorship clinics in countries with single-payer health care systems will also require stalwart leadership.16 Results such as those presented here by Sutradhar et al13 will go a long way in supporting this process.
Author Manuscript
The economic implications of a potential reduction in ED visits among adult survivors of childhood cancer cannot be ignored. One hypothesis is that observed ED visits were reduced because survivors with a history of a survivorship clinic visit sought care in an outpatient setting rather than the ED. However, the analysis was adjusted for primary care services (annual physical examinations) and continuity of care with a single provider. In addition, it does not seem likely that high-acuity visits or ED-initiated hospitalizations, which were also reduced among those with a survivorship clinic visit, could be replaced in this way. Finally, regardless of whether underlying morbidity was reduced, this transfer of services could also be expected to reduce health care costs.17 Health care delivery researchers should also take note of Sutradhar et al's article13 with respect to the study design. Investigators used multiple linked administrative health databases to take advantage of a local policy decision: the institution of specialty survivorship clinics by the Ontario Ministry of Health and Long-Term Care. Because of limitations of the available data, these investigators could not use a before/after study design. Nonetheless, should a similar initiative take place elsewhere, the effects could be examined as a natural experiment.18
Author Manuscript Author Manuscript
Finally, the clinical care implications of Sutradhar et al's findings13 must be acknowledged. Although underlying morbidity could not be examined, a substantial and significant reduction in subsequent ED visits after 1 or more survivorship clinic visits was reported. The analysis included a multivariate adjustment for cancer diagnosis, relapse, treatment characteristics (including treatment intensity), and demographic and socioeconomic variables. Therefore, although it is tempting to hypothesize that the observed associations are due to bias (sicker patients, who are more likely to visit the ED, did not attend survivor clinics), the strength of the findings after the multivariate analysis argues against this possibility. Therefore, we must ask whether care for adult survivors of childhood cancer is so specialized that it requires the contributions of a dedicated provider. As noted by the authors, such specialized clinics are intended not to replace care provided by a primary care clinician but rather to complement such care through a shared-care model.19 The report from Sutradhar et al in this issue of Cancer makes a robust argument for this strategy. So, what are the next steps? First, further study is necessary to determine whether specialized survivorship clinics reduce hospitalization rates and indeed whether they lead to a reduction in serious morbidity and an increase in long-term survival. The design of such studies is complex; we encourage the use of natural experiments described previously and similar registry data-linkage studies. We also posit that many adult survivors of childhood cancer do not need specialized services because the risk of serious late effects associated
Cancer. Author manuscript; available in PMC 2016 March 21.
Tonorezos and Oeffinger
Page 3
Author Manuscript
with some cancer therapies is rather modest. Edgar et al,20 extending previous work by Wallace et al,21 developed a simple method for predicting long-term outcomes and stratifying risk into 3 levels based on treatment exposures. Use of this tool should help to distinguish who might benefit from care in specialized survivorship clinics from those survivors who would simply be overmedicalized. Lastly, 1 population of cancer survivors in Sutradhar et al's study13 appeared to have a disproportionately low percentage of visits to the survivor clinics: central nervous system tumor survivors. In comparison with other cancer groups, survivors of a childhood central nervous system tumor have a substantially higher risk of having cognitive dysfunction and problems with sight, hearing, and balance and thus often become socially isolated.22 Thus, it is imperative that methods be developed and tested with the intent of minimizing the loss to follow-up of this particularly high-risk population.
Acknowledgments Author Manuscript
Funding Support: This work was supported in part by the National Institutes of Health (K05 CA160724 and R01 CA134722 to Kevin C. Oeffinger [principal investigator]) R01 CA187397 to Emily S. Tonorezos [principal investigator]).
References
Author Manuscript Author Manuscript
1. Smith MA, Reaman GH. Remaining challenges in childhood cancer and newer targeted therapeutics. Pediatr Clin North Am. 2015; 62:301–312. [PubMed: 25435124] 2. Mukherjee, S. The Emperor of All Maladies: A Biography of Cancer. New York, NY: Simon & Schuster; 2011. 3. Hudson MM, Neglia JP, Woods WG, et al. Lessons from the past: opportunities to improve childhood cancer survivor care through outcomes investigations of historical therapeutic approaches for pediatric hematological malignancies. Pediatr Blood Cancer. 2012; 58:334–343. [PubMed: 22038641] 4. Robison LL, Hudson MM. Survivors of childhood and adolescent cancer: life-long risks and responsibilities. Nat Rev Cancer. 2014; 14:61–70. [PubMed: 24304873] 5. Oeffinger KC, Mertens AC, Sklar CA, et al. Chronic health conditions in adult survivors of childhood cancer. N Engl J Med. 2006; 355:1572–1582. [PubMed: 17035650] 6. Armstrong GT, Kawashima T, Leisenring W, et al. Aging and risk of severe, disabling, lifethreatening, and fatal events in the Childhood Cancer Survivor Study. J Clin Oncol. 2014; 32:1218– 1227. [PubMed: 24638000] 7. Landier W, Bhatia S, Eshelman DA, et al. Development of risk-based guidelines for pediatric cancer survivors: the Children's Oncology Group Long-Term Follow-Up Guidelines from the Children's Oncology Group Late Effects Committee and Nursing Discipline. J Clin Oncol. 2004; 22:4979– 4990. [PubMed: 15576413] 8. Children's Oncology Group. [Accessed August 2015.] Long-term follow-up guidelines for survivors of childhood, adolescent, and young adult cancers. http://www.survivorshipguidelines.org/ 9. American Society of Clinical Oncology. [Accessed August 2015.] Workforce initiatives. http:// www.asco.org/practice-research/workforce-initiatives 10. Tonorezos ES, Henderson TO. Clinical guidelines for the care of childhood cancer survivors. Children. 2014; 1:227–240. 11. Suh E, Daugherty CK, Wroblewski K, et al. General internists' preferences and knowledge about the care of adult survivors of childhood cancer: a cross-sectional survey. Ann Intern Med. 2014; 160:11–17. [PubMed: 24573662] 12. Nathan PC, Daugherty CK, Wroblewski KE, et al. Family physician preferences and knowledge gaps regarding the care of adolescent and young adult survivors of childhood cancer. J Cancer Surviv. 2013; 7:275–282. [PubMed: 23471729]
Cancer. Author manuscript; available in PMC 2016 March 21.
Tonorezos and Oeffinger
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Author Manuscript Author Manuscript
13. Sutradhar R, Agha M, Pole JD, et al. Specialized survivor clinic attendance is associated with decreased rates of emergency department visits in adult survivors of childhood cancer. Cancer. 2015; 121 14. Oeffinger KC, Argenbright KE, Levitt GA, et al. Models of cancer survivorship health care: moving forward. Am Soc Clin Oncol Educ Book. 2014:205–213. [PubMed: 24857078] 15. Centers for Medicare & Medicaid Services. [Accessed 9, 2014] Medicare approved facilities/trials/ registries. http://www.cms.gov/Medicare/Medicare-General-Information/ MedicareApprovedFacilitie/index.html 16. Jefford M, Rowland J, Grunfeld E, Richards M, Maher J, Glaser A. Implementing improved posttreatment care for cancer survivors in England, with reflections from Australia, Canada and the USA. Br J Cancer. 2013; 108:14–20. [PubMed: 23257892] 17. Mehrotra A, Liu H, Adams JL, et al. Comparing costs and quality of care at retail clinics with that of other medical settings for 3 common illnesses. Ann Intern Med. 2009; 151:321–328. [PubMed: 19721020] 18. Craig P, Cooper C, Gunnell D, et al. Using natural experiments to evaluate population health interventions: new Medical Research Council guidance. J Epidemiol Community Health. 2012; 66:1182–1186. [PubMed: 22577181] 19. Oeffinger KC, McCabe MS. Models for delivering survivorship care. J Clin Oncol. 2006; 24:5117–5124. [PubMed: 17093273] 20. Edgar AB, Duffin K, Borthwick S, Marciniak-Stepak P, Wallace WH. Can intensity of long-term follow-up for survivors of childhood and teenage cancer be determined by therapy-based risk stratification? BMJ Open. 2013; 3:e002451. 21. Wallace WH, Blacklay A, Eiser C, et al. Developing strategies for long term follow up of survivors of childhood cancer. BMJ. 2001; 323:271–274. [PubMed: 11485960] 22. Vannatta K, Gerhardt CA, Wells RJ, Noll RB. Intensity of CNS treatment for pediatric cancer: prediction of social outcomes in survivors. Pediatr Blood Cancer. 2007; 49:716–722. [PubMed: 17096410]
Author Manuscript Author Manuscript Cancer. Author manuscript; available in PMC 2016 March 21.
Cancer. Author manuscript; available in PMC 2016 March 21. Published in final edited form as: Cancer. 2015 December 15; 121(24): 4279–4281. doi:10.1002/cncr.29683.
Specialty Care for Adult Survivors of Childhood Cancer Emily S. Tonorezos, MD, MPH and Kevin C. Oeffinger, MD Department of Medicine, Memorial Sloan Kettering Cancer Center, New York, New York
Author Manuscript
The field of pediatric oncology has made enormous advances over the past decades, as evidenced in countless scholarly publications and the lay press.1-3 Among the primary beneficiaries of these successes are the more than 400,000 adult survivors of childhood cancer currently living in the United States.4 Unfortunately, late effects of childhood cancer therapy are common, serious, and potentially life-threatening.5,6 Recommended screening and surveillance for late effects are carefully outlined in the Children's Oncology Group guidelines,7,8 but the pool of available oncologists is shrinking,9 and primary care providers express uncertainty regarding how best to care for survivors.10-12 Furthermore, a fragmented approach to health care delivery in the United States results in a haphazard and nonsystematic approach to care for this high-risk population.
Author Manuscript Author Manuscript
In this issue of Cancer, Sutradhar et al13 from Canada have taken advantage of the visionary 1999 decision by the Ontario Ministry of Health and Long-Term Care to initiate a provincial network of specialty clinics for adult survivors of childhood cancer. Using multiple linked administrative health databases, the authors created a population-based cohort of 3912 adult survivors of childhood cancer diagnosed between 1986 and 2005 in Ontario, Canada. Approximately 43% of this cohort attended at least 1 survivor clinic visit during the study period, and 14,187 emergency department (ED) visits were captured. An Andersen-Gill recurrent event multivariate regression model was used, and the results demonstrated that 1 or more prior visits to a survivor clinic was associated with a 19% lower rate of ED visits in comparison with survivors who had not visited a specialty survivor clinic. When a visit to a survivor clinic was considered a cumulative count covariate, each additional visit to a survivor clinic decreased the subsequent ED visit rate by 5%. In further analysis, these results were independent of whether or not the survivor was also seen by a primary care physician, and importantly, both low-acuity and high-acuity ED visits were reduced. On the basis of the strength of these findings, it is reasonable to state that attendance at a specialized survivor clinic results in fewer ED visits; whether underlying morbidity is also affected is an intriguing possibility. The policy, economic, and clinical care implications of these findings are significant. The Ontario Ministry of Health and Long-Term Care made the prescient decision to support the creation of 5 specialty clinics for adult survivors of childhood cancer in 1999. In the United States, a small number of cancer center–based programs for adult survivors of childhood
Corresponding author: Kevin C. Oeffinger, MD, Adult Long-Term Follow-Up Program, Memorial Sloan Kettering Cancer Center, 485 Lexington Avenue, 2nd Floor, New York, NY 10017; Fax: (214) 648-2551; [email protected]. Conflict of Interest Disclosures: The authors made no disclosure.
Tonorezos and Oeffinger
Page 2
Author Manuscript
cancer exist, but their reach is limited, and reimbursement for services can be difficult.14 Reducing ED visits for survivors could be achieved with an expansion of specialty clinics in the United States, perhaps via a Medicare approval process, as is already in place for intensive cardiac rehabilitation programs and other facities.15 though it could be less problematical, establishing survivorship clinics in countries with single-payer health care systems will also require stalwart leadership.16 Results such as those presented here by Sutradhar et al13 will go a long way in supporting this process.
Author Manuscript
The economic implications of a potential reduction in ED visits among adult survivors of childhood cancer cannot be ignored. One hypothesis is that observed ED visits were reduced because survivors with a history of a survivorship clinic visit sought care in an outpatient setting rather than the ED. However, the analysis was adjusted for primary care services (annual physical examinations) and continuity of care with a single provider. In addition, it does not seem likely that high-acuity visits or ED-initiated hospitalizations, which were also reduced among those with a survivorship clinic visit, could be replaced in this way. Finally, regardless of whether underlying morbidity was reduced, this transfer of services could also be expected to reduce health care costs.17 Health care delivery researchers should also take note of Sutradhar et al's article13 with respect to the study design. Investigators used multiple linked administrative health databases to take advantage of a local policy decision: the institution of specialty survivorship clinics by the Ontario Ministry of Health and Long-Term Care. Because of limitations of the available data, these investigators could not use a before/after study design. Nonetheless, should a similar initiative take place elsewhere, the effects could be examined as a natural experiment.18
Author Manuscript Author Manuscript
Finally, the clinical care implications of Sutradhar et al's findings13 must be acknowledged. Although underlying morbidity could not be examined, a substantial and significant reduction in subsequent ED visits after 1 or more survivorship clinic visits was reported. The analysis included a multivariate adjustment for cancer diagnosis, relapse, treatment characteristics (including treatment intensity), and demographic and socioeconomic variables. Therefore, although it is tempting to hypothesize that the observed associations are due to bias (sicker patients, who are more likely to visit the ED, did not attend survivor clinics), the strength of the findings after the multivariate analysis argues against this possibility. Therefore, we must ask whether care for adult survivors of childhood cancer is so specialized that it requires the contributions of a dedicated provider. As noted by the authors, such specialized clinics are intended not to replace care provided by a primary care clinician but rather to complement such care through a shared-care model.19 The report from Sutradhar et al in this issue of Cancer makes a robust argument for this strategy. So, what are the next steps? First, further study is necessary to determine whether specialized survivorship clinics reduce hospitalization rates and indeed whether they lead to a reduction in serious morbidity and an increase in long-term survival. The design of such studies is complex; we encourage the use of natural experiments described previously and similar registry data-linkage studies. We also posit that many adult survivors of childhood cancer do not need specialized services because the risk of serious late effects associated
Cancer. Author manuscript; available in PMC 2016 March 21.
Tonorezos and Oeffinger
Page 3
Author Manuscript
with some cancer therapies is rather modest. Edgar et al,20 extending previous work by Wallace et al,21 developed a simple method for predicting long-term outcomes and stratifying risk into 3 levels based on treatment exposures. Use of this tool should help to distinguish who might benefit from care in specialized survivorship clinics from those survivors who would simply be overmedicalized. Lastly, 1 population of cancer survivors in Sutradhar et al's study13 appeared to have a disproportionately low percentage of visits to the survivor clinics: central nervous system tumor survivors. In comparison with other cancer groups, survivors of a childhood central nervous system tumor have a substantially higher risk of having cognitive dysfunction and problems with sight, hearing, and balance and thus often become socially isolated.22 Thus, it is imperative that methods be developed and tested with the intent of minimizing the loss to follow-up of this particularly high-risk population.
Acknowledgments Author Manuscript
Funding Support: This work was supported in part by the National Institutes of Health (K05 CA160724 and R01 CA134722 to Kevin C. Oeffinger [principal investigator]) R01 CA187397 to Emily S. Tonorezos [principal investigator]).
References
Author Manuscript Author Manuscript
1. Smith MA, Reaman GH. Remaining challenges in childhood cancer and newer targeted therapeutics. Pediatr Clin North Am. 2015; 62:301–312. [PubMed: 25435124] 2. Mukherjee, S. The Emperor of All Maladies: A Biography of Cancer. New York, NY: Simon & Schuster; 2011. 3. Hudson MM, Neglia JP, Woods WG, et al. Lessons from the past: opportunities to improve childhood cancer survivor care through outcomes investigations of historical therapeutic approaches for pediatric hematological malignancies. Pediatr Blood Cancer. 2012; 58:334–343. [PubMed: 22038641] 4. Robison LL, Hudson MM. Survivors of childhood and adolescent cancer: life-long risks and responsibilities. Nat Rev Cancer. 2014; 14:61–70. [PubMed: 24304873] 5. Oeffinger KC, Mertens AC, Sklar CA, et al. Chronic health conditions in adult survivors of childhood cancer. N Engl J Med. 2006; 355:1572–1582. [PubMed: 17035650] 6. Armstrong GT, Kawashima T, Leisenring W, et al. Aging and risk of severe, disabling, lifethreatening, and fatal events in the Childhood Cancer Survivor Study. J Clin Oncol. 2014; 32:1218– 1227. [PubMed: 24638000] 7. Landier W, Bhatia S, Eshelman DA, et al. Development of risk-based guidelines for pediatric cancer survivors: the Children's Oncology Group Long-Term Follow-Up Guidelines from the Children's Oncology Group Late Effects Committee and Nursing Discipline. J Clin Oncol. 2004; 22:4979– 4990. [PubMed: 15576413] 8. Children's Oncology Group. [Accessed August 2015.] Long-term follow-up guidelines for survivors of childhood, adolescent, and young adult cancers. http://www.survivorshipguidelines.org/ 9. American Society of Clinical Oncology. [Accessed August 2015.] Workforce initiatives. http:// www.asco.org/practice-research/workforce-initiatives 10. Tonorezos ES, Henderson TO. Clinical guidelines for the care of childhood cancer survivors. Children. 2014; 1:227–240. 11. Suh E, Daugherty CK, Wroblewski K, et al. General internists' preferences and knowledge about the care of adult survivors of childhood cancer: a cross-sectional survey. Ann Intern Med. 2014; 160:11–17. [PubMed: 24573662] 12. Nathan PC, Daugherty CK, Wroblewski KE, et al. Family physician preferences and knowledge gaps regarding the care of adolescent and young adult survivors of childhood cancer. J Cancer Surviv. 2013; 7:275–282. [PubMed: 23471729]
Cancer. Author manuscript; available in PMC 2016 March 21.
Tonorezos and Oeffinger
Page 4
Author Manuscript Author Manuscript
13. Sutradhar R, Agha M, Pole JD, et al. Specialized survivor clinic attendance is associated with decreased rates of emergency department visits in adult survivors of childhood cancer. Cancer. 2015; 121 14. Oeffinger KC, Argenbright KE, Levitt GA, et al. Models of cancer survivorship health care: moving forward. Am Soc Clin Oncol Educ Book. 2014:205–213. [PubMed: 24857078] 15. Centers for Medicare & Medicaid Services. [Accessed 9, 2014] Medicare approved facilities/trials/ registries. http://www.cms.gov/Medicare/Medicare-General-Information/ MedicareApprovedFacilitie/index.html 16. Jefford M, Rowland J, Grunfeld E, Richards M, Maher J, Glaser A. Implementing improved posttreatment care for cancer survivors in England, with reflections from Australia, Canada and the USA. Br J Cancer. 2013; 108:14–20. [PubMed: 23257892] 17. Mehrotra A, Liu H, Adams JL, et al. Comparing costs and quality of care at retail clinics with that of other medical settings for 3 common illnesses. Ann Intern Med. 2009; 151:321–328. [PubMed: 19721020] 18. Craig P, Cooper C, Gunnell D, et al. Using natural experiments to evaluate population health interventions: new Medical Research Council guidance. J Epidemiol Community Health. 2012; 66:1182–1186. [PubMed: 22577181] 19. Oeffinger KC, McCabe MS. Models for delivering survivorship care. J Clin Oncol. 2006; 24:5117–5124. [PubMed: 17093273] 20. Edgar AB, Duffin K, Borthwick S, Marciniak-Stepak P, Wallace WH. Can intensity of long-term follow-up for survivors of childhood and teenage cancer be determined by therapy-based risk stratification? BMJ Open. 2013; 3:e002451. 21. Wallace WH, Blacklay A, Eiser C, et al. Developing strategies for long term follow up of survivors of childhood cancer. BMJ. 2001; 323:271–274. [PubMed: 11485960] 22. Vannatta K, Gerhardt CA, Wells RJ, Noll RB. Intensity of CNS treatment for pediatric cancer: prediction of social outcomes in survivors. Pediatr Blood Cancer. 2007; 49:716–722. [PubMed: 17096410]
Author Manuscript Author Manuscript Cancer. Author manuscript; available in PMC 2016 March 21.
