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Journal of Clinical Oncology, Vol 26, No 27 (September 20), 2008: pp. 4401-4409 © 2008 American Society of Clinical Oncology. DOI: 10.1200/JCO.2008.16.9607 Medical Care in Long-Term Survivors of Childhood Cancer: A Report From the Childhood Cancer Survivor Study
From the Hospital for Sick Children, Toronto, Ontario, Canada; St Jude Children's Research Hospital, Memphis, TN; Emory University, Atlanta, GA; Roswell Park Cancer Institute, Buffalo, NY; University of Michigan, Ann Arbor, MI; Stanford Medical Center, Stanford, CA; Fred Hutchison Cancer Research Center, Seattle, WA; and Memorial Sloan-Kettering Cancer Center, New York, NY Corresponding author: Paul Nathan, MD, MSc, the Hospital for Sick Children, Division of Haematology/Oncology, 555 University Ave, Toronto, ON M5G 1X8, Canada, e-mail: paul.nathan{at}sickkids.ca
Purpose To evaluate whether childhood cancer survivors receive regular medical care focused on the specific morbidities that can arise from their therapy. Patients and Methods We conducted a cross-sectional survey of health care use in 8,522 participants in the Childhood Cancer Survivor Study, a multi-institutional cohort of childhood cancer survivors. We assessed medical visits in the preceding 2 years, whether these visits were related to the prior cancer, whether survivors received advice about how to reduce their long-term risks, and whether screening tests were discussed or ordered. Completion of echocardiograms and mammograms were assessed in patients at high risk for cardiomyopathy or breast cancer. We examined the relationship between demographics, treatment, health status, chronic medical conditions, and health care use. Results Median age at cancer diagnosis was 6.8 years (range, 0 to 20.9 years) and at interview was 31.4 years (range, 17.5 to 54.1 years). Although 88.8% of survivors reported receiving some form of medical care, only 31.5% reported care that focused on their prior cancer (survivor-focused care), and 17.8% reported survivor-focused care that included advice about risk reduction or discussion or ordering of screening tests. Among survivors who received medical care, those who were black, older at interview, or uninsured were less likely to have received risk-based, survivor-focused care. Among patients at increased risk for cardiomyopathy or breast cancer, 511 (28.2%) of 1,810 and 169 (40.8%) of 414 had undergone a recommended echocardiogram or mammogram, respectively. Conclusion Despite a significant risk of late effects after cancer therapy, the majority of childhood cancer survivors do not receive recommended risk-based care.
With contemporary therapy, 80% of children diagnosed with cancer will become long-term survivors.1 Consequently, more than 270,000 survivors of childhood cancer are alive in the United States,2 many of whom are at risk of long-term morbidity3,4 and premature mortality5,6 as a result of their therapy. We have estimated that by 30 years from their cancer diagnosis, 73% will develop at least one chronic physical health condition and 42% will develop a severe, life-threatening, or disabling condition or die from a chronic condition.4 Compared with their siblings, survivors are 10 times more likely to develop a serious chronic disease (eg, second cancer or heart disease), and the risk of morbidity and premature mortality does not plateau.4 Because the risk for many serious health problems can be reduced by prevention or early detection, the Institute of Medicine (IOM) has recommended lifelong risk-based health care for all cancer survivors.2 This requires a systematic plan for periodic screening, surveillance, and prevention that is adapted to the risks arising from the previous cancer, its therapy, genetic predispositions, lifestyle behaviors, and comorbid health conditions.2,7 The frequency and intensity of surveillance should be adapted to each survivor's risk, with those at low risk for sequelae requiring less contact and those at significantly increased risk of morbidity or premature mortality (eg, those treated with radiation therapy or stem-cell transplantation) requiring annual monitoring. The purpose of this study was to characterize the patterns and predictors of health care use among a large cohort of geographically and socioeconomically diverse childhood cancer survivors. In particular, we were interested in determining the proportion of individuals who received medical care that was focused specifically on addressing and reducing the risks arising from their prior cancer therapy.
Childhood Cancer Survivor Study The Childhood Cancer Survivor Study (CCSS) methodology and a description of the participants have been published.8 Briefly, the cohort includes individuals diagnosed with cancer before age 21 years at one of 26 centers (25 centers in the United States and one in Canada) from 1970 to 1986 who were alive at least 5 years from their original diagnosis. The eligible cohort consisted of 20,720 patients; 17,703 were successfully contacted and 14,366 (81.2%) enrolled in the study. There were no significant differences between participants and nonparticipants by sex, age at diagnosis or at cohort assembly, cancer type, or treatment.8,9 Detailed medical information was abstracted from participants’ hospital records. Participants completed a comprehensive baseline questionnaire and several subsequent questionnaires. Eligibility for this analysis was limited to participants who completed a questionnaire in 2002 to 2003 that addressed health care use and for whom information regarding treatment for their original cancer was available. Questionnaires and data abstraction forms are available at www.stjude.org/ccss. The study was approved by the institutional review board at each participating institution, and informed consent was obtained from each participant or parent/guardian.
Health Care Use
Predictors of Health Care Use Chronic medical conditions and health status. We evaluated the association between health status, chronic medical conditions, and health care use. The severity of chronic health conditions reported on the baseline questionnaire was classified as (1) mild, (2) moderate, (3) severe, or (4) life-threatening or disabling, using the National Cancer Institute's Common Terminology Criteria for Adverse Events (version 3), as published previously.4 Health status was measured on the 2002 to 2003 questionnaire using a previously defined set of domains (emotional health,13,14 physical functioning,15 cancer-related pain, and cancer-related anxiety and fears14).
Statistical Analysis
Characteristics of the Study Cohort Of the 14,366 survivors who were enrolled in the CCSS, 1,773 did not have medical records available, 1,017 were dead at the time of the baseline assessment and had their questionnaire completed by a proxy respondent, and 462 died before the mailing out of the 2002 to 2003 questionnaire. Thus 11,114 survivors were eligible for this study of health care use. Of these, 1,919 (17.3%) were nonrespondents and 673 (6.1%) were lost to follow-up, resulting in 8,522 (76.7%) survivors being available for this analysis. The 8,522 participants did not differ from the 2,592 eligible nonparticipants by diagnosis, therapy, or current age, but were more likely to be female (79.8% of eligible females v 73.9% of eligible males; P < .001) and older when diagnosed with their original cancer (mean age, 8.2 v 7.5 years; P = .006). Demographic, treatment, and health status characteristics of the participants are presented in Table 1.
Health Care Use During the 2-year study period, 953 survivors (11.2%) reported receiving no medical care, 4,882 survivors (57.3%) reported receiving general medical care, 1,166 survivors (13.7%) reported receiving general survivor-focused care, and 1,521 survivors (17.8%) reported receiving risk-based, survivor-focused care. Table 1 lists the percentage of survivors who reported each level of care by demographic, treatment, and health status variables. Only 1,246 patients (14.6%) received care at a cancer center. The probability of receiving a particular level of care according to cancer diagnosis is shown in Figure 1. General survivor-focused or risk-based survivor-focused care was most likely among survivors of CNS tumors. Survivors of neuroblastoma were most likely to have received no medical care.
The relationship between demographic and health status variables and the odds of having received no medical care is summarized in Table 2. Male survivors, uninsured survivors, and those with a household income less than $40,000/year were more likely to report no medical visits. In contrast, survivors who reported moderate to extreme cancer-related pain were more likely to have received care, as were those with a severe, life-threatening, or disabling chronic health condition. Age at diagnosis and at interview were not associated with having received medical care.
Additionally, Table 2 presents predictors of reporting general medical care rather than risk-based, survivor-focused care among survivors who reported some form of care (n = 7,569). Survivors who were older, male, black, or uninsured were more likely to report general care rather than risk-based, survivor-focused care. In contrast, survivors who reported moderate to extreme cancer-related pain or anxiety, poor physical health, or more serious morbidity were more likely to report risk-based, survivor-focused care. Among survivors who received some form of survivor-focused care, only the location of care (cancer center v other location; odds ratio = 1.52, 95% CI, 1.29 to 1.80) and "other" race (v white race; odds ratio = 1.38, 95% CI, 1.05 to 1.84) influenced whether this care was risk-based. The relationship between therapeutic exposures associated with an increased risk of late effects and the level of care is summarized in Table 3. Patients treated with an alkylating agent or anthracycline (without chest radiation), two therapies that are strongly associated with long-term morbidity, were no more likely to report receiving any health care rather than no health care. Similarly, survivors who received an anthracycline (without chest radiation) were no more likely to receive risk-based, survivor-focused care than general care, although risk-based, survivor-focused care was more likely in survivors who received the highest tertile of alkylating agent dosing. In contrast, all survivors treated with radiation therapy were more likely to report risk-based, survivor-focused care.
An echocardiogram was indicated in 1,810 survivors and a mammogram was indicated in 414 women based on their high risk for developing a cardiomyopathy or breast cancer, respectively. However, only 511 (28.2%) of 1,810 survivors at risk for cardiomyopathy reported an echocardiogram. For women treated with chest radiation who were 27 years or older at the time of the 2002 to 2003 questionnaire and were younger than the age at which routine screening mammography is recommended for the general population (United States, age 40 years; Canada, age 50 years), 169 (40.8%) of 414 survivors reported receiving a mammogram. Survivors who were uninsured, Canadian, or not seen at a cancer center were more likely to not report an indicated echocardiogram, whereas those who had moderate to extreme anxiety after their cancer were more likely to have received an echocardiogram (Table 4). Only care outside of a cancer center was associated with not having received an indicated mammogram. Survivors who received care in a cancer center were more likely than those who received their care elsewhere to report an indicated echocardiogram (53.2% v 22.3%) or mammogram (62.4% v 34.6%).
Only 17.8% of this cohort of 8,522 long-term survivors of childhood cancer reported a medical visit within the previous 2 years during which their health care provider specifically addressed the risks arising from their therapy. This low prevalence of risk-based care falls far below the goals advocated by the IOM.2 Strikingly, 88.8% of survivors had at least one medical visit during the study period, reflecting that access to medical care was not a barrier for most. Rather, the care that they received did not focus on their specific risks and strategies to ameliorate them. Fewer than 15% of survivors received medical care at a cancer center; most received their care from a primary care clinician in a community setting. However, survivors cared for in the community were less likely to report risk-based, survivor-focused care or to have received indicated echocardiography or mammography. Because more than 70% of survivors will develop one or more chronic conditions related to their prior therapy,3,4 this low rate of risk-based, survivor-focused care suggests multiple lost opportunities to prevent or expeditiously detect and treat these sequelae. Thus efforts must be focused on providing primary care clinicians with the education and resources needed to provide risk-based care to this group of patients.17 Previous studies have shown that key barriers to appropriate survivor care include a lack of patient and physician knowledge about the long-term risks of cancer therapy.18,19 Survivors are often unaware of the details of their cancer therapy, preventing them from seeking care focused on specific risks. One study has reported that among exposed survivors, fewer than one third recalled receiving anthracycline chemotherapy, which is associated with a risk of late cardiac toxicity.20,21 Similar knowledge deficits are likely in our study, as survivors who received anthracyclines were no more likely to get care than those who had not received anthracyclines. Only survivors who had received chest radiation were more likely to receive such care, suggesting that care-seeking behavior among those at risk for cardiac disease is influenced by radiation exposure, but not anthracycline therapy. This observation is particularly concerning given that approximately half of children treated for cancer will receive anthracycline chemotherapy. Similarly, survivors treated with low or moderate doses of alkylating agents, which have been associated with secondary leukemias22 and infertility,23-25 were no more likely to receive care than those not treated with these drugs. Forty-one percent of females at increased risk for breast cancer reported a mammogram within the prior 2 years, despite evidence that by 45 years of age, 20% of women treated with chest radiation as children will develop breast cancer.26 Only 28% of survivors at risk for a cardiomyopathy reported an echocardiogram. Self-report of mammography has been demonstrated to be valid in several studies, with 81% to 97% congruence between patient self-report and medical record audits.27-30 There are no published data on the validity of self-report of echocardiograms. Although patients who received their care at a cancer center had higher rates of appropriate surveillance, many patients treated at these specialized centers did not undergo the recommended tests. Whether the failure to perform a recommended echocardiogram in 47% and a mammogram in 38% of patients seen at a cancer center was due to lack of insurance coverage, inadequate physician knowledge, or an active decision not to heed the guidelines cannot be deduced from this study. The provision of a comprehensive survivorship care plan to all survivors, as recommended by the IOM,2,7 may increase the number of survivors who receive recommended surveillance tests such as echocardiography or breast imaging. As noted in studies of health care use in other diseases, male patients, the uninsured, and those with lower household incomes31 are particularly vulnerable, because they are at greater risk of receiving no medical care at all. Almost 30% of uninsured survivors had not received medical care in the previous 2 years, compared with 10% of insured survivors. In comparison, in a study of 1,718 survivors of adult cancer that used data from the National Health Interview Survey (1998 and 2000 surveys), 45.1% of uninsured survivors reported not getting needed medical care within the preceding year because of concerns about the cost of that care; in contrast, 16.7% of publicly insured and 4.4% of privately insured survivors did not receive needed care.32 Among survivors in our study who reported some form of care, those who had developed sequelae of their prior therapy (such as pain, anxiety, poorer physical health, or a severe chronic physical condition) were more likely to report a visit related to their previous malignancy, suggesting that these individuals may have been seeking care for extant symptoms. However, because the incidence of serious chronic health conditions increases as survivors age,3,4 even survivors who have not developed late effects may benefit from care focused on prevention and early detection.17 Several methodologic limitations should be considered when interpreting the results. First, data were obtained from self-reports. We cannot examine whether survivors’ impressions about the purpose and content of their medical visit was concordant with their caregivers’ intentions. Second, the results are derived from 8,522 of 11,114 eligible survivors. This potential for selection bias is compounded by the observation that CCSS participants are a select group of survivors, likely better educated about the potential late effects of their cancer therapy than nonparticipants. Thus this study likely overestimates the proportion of survivors who receive appropriate care, and the poor compliance with recommended care demonstrated here is probably a best-case scenario. Third, this cohort of survivors received their therapy from 1970 to 1986. Caution should be exercised in generalizing to patients treated more recently. It is plausible that patients treated in the current era are better informed about the long-term risks of their therapy. The CCSS is currently recruiting a cohort of survivors treated from 1987 to 1999 to examine such questions. In summary, despite a significant risk of late effects after cancer therapy, the majority of adult survivors of childhood cancer do not receive regular medical care focused on their long-term risks. Only a minority of survivors at the highest risk for developing cardiac dysfunction or breast cancer receive recommended surveillance tests. The majority of survivors receive medical care in a community setting where, they are less likely to receive risk-based care or recommended screening tests. Given the rapidly expanding population of childhood cancer survivors, it is imperative that efforts be focused on educating both survivors and the health care providers who will care for them as adults about the importance of regular, risk-based medical care.
The author(s) indicated no potential conflicts of interest.
Conception and design: Paul C. Nathan, Mark L. Greenberg, Kirsten K. Ness, Melissa M. Hudson, James G. Gurney, Sarah S. Donaldson, Leslie L. Robison, Kevin C. Oeffinger Administrative support: Ann C. Mertens Provision of study materials or patients: Melissa M. Hudson, Ann C. Mertens Collection and assembly of data: Melissa M. Hudson, Ann C. Mertens, Sarah S. Donaldson, Wendy M. Leisenring, Leslie L. Robison Data analysis and interpretation: Paul C. Nathan, Mark L. Greenberg, Kirsten K. Ness, Melissa M. Hudson, Martin C. Mahoney, James G. Gurney, Sarah S. Donaldson, Wendy M. Leisenring, Kevin C. Oeffinger Manuscript writing: Paul C. Nathan, Mark L. Greenberg, Kirsten K. Ness, Kevin C. Oeffinger Final approval of manuscript: Paul C. Nathan, Mark L. Greenberg, Kirsten K. Ness, Melissa M. Hudson, Ann C. Mertens, Martin C. Mahoney, James G. Gurney, Sarah S. Donaldson, Wendy M. Leisenring, Leslie L. Robison, Kevin C. Oeffinger
The Childhood Cancer Survivor Study (CCSS) is a collaborative, multi-institutional project, funded as a resource by the National Cancer Institute, of individuals who survived 5 or more years after diagnosis of childhood cancer. CCSS is a retrospectively ascertained cohort of 20,720 childhood cancer survivors diagnosed before age 21 years between 1970 and 1986, in addition to approximately 4,000 siblings of survivors who serve as a control group. The cohort was assembled through the efforts of 26 participating clinical research centers in the United States and Canada. The study is currently funded by a U24 resource grant (National Cancer Institute Grant No. U24 CA55727) awarded to St Jude Children's Research Hospital. Currently, we are in the process of expanding the cohort to include an additional 14,000 childhood cancer survivors diagnosed before age 21 between 1987 and 1999. For information on how to access and use the CCSS resource, visit www.stjude.org/ccss.
Supported by Grant No. U24-CA-55727 (L.L. Robison, principal investigator) from the United States Department of Health and Human Services, funding to the University of Minnesota from the Children's Cancer Research Fund, and funding to St Jude Children's Research Hospital from the American Lebanese Syrian Associated Charities. Presented in part at the 43rd Annual Meeting of the American Society of Clinical Oncology, June 1-5, 2007, Chicago, IL. Authors’ disclosures of potential conflicts of interest and author contributions are found at the end of this article.
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Copyright © 2008 by the American Society of Clinical Oncology, Online ISSN: 1527-7755. Print ISSN: 0732-183X
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ABSTRACT
INTRODUCTION
300 mg/m2 of an anthracycline or any anthracycline dose plus chest radiation) or breast cancer (females who had received radiation to the chest and who were 
2 statistics. 
