Health Care of Young Adult Survivors of Childhood Cancer: A Report from the Childhood Cancer Survivor Study

Dr. Oeffinger’s excellent analysis of health care utilization by the CCSS cohort provides an important baseline to understanding the need to prepare survivors and family practice physicians to manage and control late effects of treatment for childhood cancer. The next step is, of course, determining how to establish and pay for systems of follow-up care, which are flexible and appropriate to the needs of individual survivors and primary care physicians.

We noted the complexity of solving this problem in Recommendation 2 of the recent IOM report on childhood cancer survivors, as Dr. Sklar notes succinctly in his present comment on the Oeffinger paper. The IOM report cites the need to create and evaluate standards and alternative models of care delivery, including collaborative practices between pediatric oncologists and primary care physicians as well as hospital-based long- term follow-up clinics.

The IOM charged the National Cancer Institute with the responsibility for assembling the necessary experts to define core elements of follow-up care and its delivery. With Dr. Oeffinger’s data indicating the inadequacy of follow-up care at least in the CCSS cohort, and, with the publication of COG’s Long-Term Follow-up Guidelines, the time is ripe to press ahead at the national level to get these tasks done. Medical, advocacy and federal leadership must make a deliberate effort to develop and implement rational strategies for comprehensive and integrated care. Otherwise, we will not move beyond restating that “survivors’ awareness must be raised” and “provider education must be promoted.”

Childhood Cancer Survivorship: Improving Care and Quality of Life. Hewitt M, Weiner S, and Simone J, eds. National Academies Press Washington D.C. 2003.

Competing interests:   None declared

This excellent paper establishes baseline information on the use of appropriate health care utilisation in young adults survivors of childhood cancer separating cancer related visits from general medical contact. The majority of the survivors were treated in the 1970.s when physicians were struggling to improve the cure rate and hence less concerned and unaware of the impact of long term sequelae. We must therefore be mindful of the historical prospective. Perhaps we should not be surprised by their findings, however, we should use them to target the groups identified as at high risk of significant problems and to continue to improve the long term surveillance of our more recent survivors to prevent them slipping through the net as their follow-up interval lengthens.

The USA and Canada does not stand alone, a population based study in the UK for patients with access to a national health service, found similar trends in use of cancer related follow-up.(1) The older the patient and further from diagnosis the less likely they were to attend for cancer related appointments. Questioning of cancer physicians revealed a significant minority discharged some patients from follow-up but the reasons were not uniform and all were discharged to primary care physicians..

This paper identifies diagnostic groups but perhaps does not highlight the plight of brain tumours survivors. The brain tumour survivors access appropriate care with difficulty and in this paper only 15% are seen in cancer centre albeit over 50% attend for cancer related problems. To clarify this issue it would have been helpful to include in the high risk treatments cranial radiation >24Gy. This treatment can cause significant morbidity and would be an indication for ongoing follow- up both for evolving /persisting endocrinopathies and second tumours in a population with neuropsychological problems

The way forward must be through effective continuing education of physicians and patients. We need to find mechanisms to provide individualised follow-up programmes based on treatment received, tumour type, age at diagnosis, gender and genetic factors. The degree of surveillance required may differ significantly from minimal contact at the patients discretion to regular attendance at a cancer centre. (2) Young adults question the need for long term surveillance.(3) Therefore these programmes must be appropriate for the patients in terms of frequency of visits, suitable personnel, the distance required to travel, take into account the young person’s perspective and most importantly contributes to improved outcomes. Lesson may be learnt from other specialties were transition of care from paediatric to adult orientated follow-up is required.

At present there are a number of international long term follow-up guidelines published. They provide a good basis for informing long term follow-up but highlight the lack of true evidence based information (category 1,2 evidence) on risks and effective methods of surveillance(4). Therefore in addition, within the programmes there must be a means of updating and communicating new information. There still remains a huge challenge to enable our patients to have the opportunity to reach their maximum potential in terms of achievements and longevity. This study is a excellent starting point and with international cooperation the task maybe made easier.

1. Taylor A, Blacklay A, Davies H, Douglas C, Jenney M, Wallace H, Levitt G Long-term follow-up of survivors of childhood cancer in the UK. Pediatr Blood Cancer. 2004;42(2):161-8.

2. Wallace WH, Blacklay A, Eiser C, Davies H, Hawkins M, Levitt G, Jenney M (2001) Developing strategies for long-term follow-up of survivors of childhood cancer BMJ 323: 271-274.

3. Eiser C, Levitt G, Leiper A, Havermans T, Donovan C. (1996) Clinic audit for long-term survivors of childhood cancer. Archives of Diseases in Childhood 75: 405-409

4. Scottish Intercollegiate Guidelines Network. Long term follow up of survivors of childhood cancer. SI GN Guideline 76. Edinburgh. SIGN: 2004.

[www.sign.ac.uk]

Competing interests:   None declared

In her comment ("Are post-traumatic stress and health care utilization related?"), Sophia K. Smith raises a noteworthy question. Her suggestion that "cancer-related post-traumatic stress has a negative impact on risk-based health care utilization in survivor populations" is a distinct possibility. This is a topic which comes up on the LTS online discussion group referred to in past comments. Dr. Greenberg ("Assuring care for childhood cancer survivors") brings up several more possibilities, which also have been topics of discussion online, and have validity.

Two of Dr. Greenberg's comments are worth further input from long- term survivors. He mentions the "small proportion lacking health insurance in this sample" in relation to barriers to access to health care. Those participating in the sample were still of young age and relatively new survivors. Our LTS discussion group membership includes survivors several decades past dx. As the years wear on and late effects increase, insurance maximums are exhausted prematurely and/or co-pays and out-of-pocket expenses for non-covered medications and procedures skyrocket. Bankruptcy becomes an unwelcome reality. And all this while fighting to have unusual and often dramatic symptoms understood for what they are.

"The failure of health care providers to meet the needs of survivors" is the more potent problem. The further survivors are from initial treatments and 'cure', the more antiquated and deadly their treatments, the greater the degree of late effects, the less documentation at hand, and the less understanding to be found in the doctor's office. If a relatively-new cancer survivor presents with newly-understood symptoms, there is a better chance of them being taken seriously. But if older survivors of childhood cancers -- in their 30's or older -- complain of various symptoms and refer to their cancer, the likelihood is great that the treatment will be based on non-cancer origins or normal aging, the complaints will be dismissed as trivial, or - as too often happens - the survivor will be ridiculed. It doesn't take many such visits to convince these survivors to stay away from the doctor's office.

Dr. Greenberg makes a statement that has become quite accurate, that these survivors "are probably their own best advocates, if armed with the necessary information about their treatment." Despite fears in the medical community of the quality of information found on the Internet, a growing number of cancer survivors are sitting at their computers, searching for answers, researching repeatedly into possible answers found, and sharing them with others who are also thirsting for this knowledge. They have more time to focus on specific research into their ailments than most doctors, have a vested interest in getting safe, accurate care, and are not as prone to following fads and gimmicks as the average patient.

Doctors everywhere need to recognize the needs of this population, embrace the challenge, and welcome the input of these special people. Only then will all the other proposals regarding their care be effective.

Competing interests:   None declared

Dr. Kevin Oeffinger and his colleagues provide a much-needed examination of the current use of health care by childhood cancer survivors. Socio-demographic and cancer-related factors that were associated with lower levels of usage include: 1) lack of health insurance; 2) lack of concern for future health; 3) male sex; and 4) age 30 years or older (in comparison to those 18 to 29 years). Cancer anxiety was measured but was not statistically associated with health care usage. However, a closer examination of this cancer-related factor (see Table 2) raises an important question: Are higher levels of cancer-related anxiety associated with lower utilization of health care services? There is little difference in the percentage of general physical exams reported by survivors across four of the five measured levels of anxiety (70-73%). Yet survivors with the highest self-reported level of anxiety report the lowest percentage of physical exams (61%). In addition, there is a ten- point difference in cancer-related visits among survivors reporting “a lot of” anxiety (59%) versus survivors reporting the highest level of anxiety (49%).

There is increasing concern that post-traumatic stress symptoms in cancer survivors could lead to an avoidance to seek medical care (1). Roughly half of the survivors studied have reported a range of cancer- related post-traumatic stress symptoms such as re-experiencing the diagnosis and/or treatment, avoiding painful reminders of cancer, and being hyper-alert, watchful or on guard (2,3). Despite the potential impact to quality of life, there is an absence of studies that have examined the association between post-traumatic stress symptoms and frequency of follow-up visits with an oncologist. It is important to note that the CCSS research team did not include a standardized measure of post -traumatic stress such as the PCL-C in their study. Therefore, we can only hypothesize that cancer-related post-traumatic stress has a negative impact on risk-based health care utilization in survivor populations. Future studies that examine the relationship between post-traumatic stress and health care utilization are needed to test this hypothesis.

(1) Hobbie, W.L., Stuber, M., Meeske, K., Wissler, K., Rourke, M.T., Ruccione, K., Hinkle, A., & Kazak, A.E. (2000). Symptoms of posttraumatic stress in young adult survivors of childhood cancer. Journal of Clinical Oncology, 18(24), 4060-6. (2) Alter, C.L., Pelcovitz, D., Axelrod, A., Goldenberg, B., Harris, H., Meyers, B., Grobois, B., Mandel, F., Septimus, A., & Kaplan, S. (1996). The identification of PTSD in cancer survivors. Psychosomatics, 37, 137-143. (3) Cordova, M.J., Andrykowski, M.A., Redd, W.H., Kenady, D.E., McGrath, P.C., & Sloan, D.A. (1995). Frequency and correlates of posttraumatic stress disorder-like symptoms after treatment for breast cancer. Journal of Consultative Clinical Psychology, 63, 981-986.

Competing interests:   None declared

I am a 32-year survivor of Wilms' tumor, which metastasized to my lung. I am also a member of the Long-Term Survivors (LTS) online discussion list mentioned by Linda Goettina.

I was very pleased to read the article by Kevin Oeffinger, et al, Health Care of Young Adult Survivors of Childhood Cancer: A Report from the Childhood Cancer Survivor Study. This article notes, “there has not been a national effort to foster linkages between childhood cancer centers and primary care physicians to enhance risk-based care.” I would like to underscore this point with my own experience. Though I have a supportive primary care provider and clearly benefit from the work done by groups like the Children’s Oncology Group (COG), it is safe to say that the only reason I actually receive adequate care and screening as a long-term survivor of childhood cancer is due to my own efforts. I would not be where I am today if I had not taken the step to begin (and maintain) the necessary research and collection of information regarding long-term survivorship.

The first of many serious “late effects” of treatment was diagnosed when I was nine years old. However, it was only two years ago that I decided to really research these issues. With much digging I found that important resources exist and discovered a whole community of individuals facing similar problems. Now I go to my doctor armed with books, the Children’s Oncology Group guidelines, and other resources and rest easier knowing that I am doing everything I can to prolong my life and ensure the best quality of life.

Here are the five ways I believe that physicians, survivors and other stakeholders can work together to help childhood cancer survivors lead better and longer lives.

1. Raise Awareness among Medical Professionals

Articles like this one are certainly a step in the right direction, but there is much yet to be done. I understand that providers do not see long- term cancer survivors every day. Yet health professionals are trained to recognize and treat many rare conditions. So many of us are labeled hypochondriacs because we have a condition that is difficult to diagnose, or drug seekers because we have pain, or are misdiagnosed because no one so young could possibly have the type of problems we encounter. This is a lack of information, but is also partially a problem of perception, the perception that the cancer “cure” is the end of a story rather than the beginning of another chapter. As noted in last year’s IOM report Childhood Cancer Survivorship: Improving Care and Quality of Life, “late effects have emerged among subgroups of childhood cancer survivors that have contributed to an appreciation of cancer as a chronic disease with implications for continuing care.”

2. Raise Awareness among Childhood Cancer Survivors

A recent JAMA article by Nancy S. Kadan-Lottick, MD, et al (Childhood Cancer Survivors’ Knowledge About Their Past Diagnosis and Treatment: Childhood Cancer Survivor Study - JAMA, April 10, 2002, Vol. 287, No. 14; pp. 1832-1839) showed that most childhood cancer survivors know precious little about their medical history. As has been noted, survivors must be helped to understand their medical past, present and future. To focus this education on today’s young children with cancer and their families is just a start and would be unacceptable as a single strategy.

3. Modify Rules for Disability Programs

Not all of the two-thirds majority of childhood cancer survivors will have late effects that disable, but some will. When I applied for Supplemental Security Income (SSI) in it took a full year to determine eligibility. While I spent the better part of a year hospitalized, the Social Security office deliberated about whether or not I was really fit for full time work. I have found that this experience is not unusual. Disability programs need to be updated to acknowledge the debilitating late effects of cancer treatment. In the meantime, primary care providers can be educated to be knowledgeable allies in the application process.

4. Increase Research Projects Related to Long-Term Survivorship

The Institute of Medicine report states that “Only systematic follow- up of large cohorts of survivors can reveal the full extent of late effects. Amelioration of these effects will require investments in intervention and clinical research to find targeted therapies that maximize survival and minimize late effects.” The cancer survivors I speak with are honored to participate in longitudinal research studies. These research projects need to be enhanced and prolonged to capture information throughout the survivor’s life span so that late effects not yet understood are discovered and can be dealt with appropriately and proactively.

5. Increase Health Insurance Accessibility

Uninsured people are forced to delay treatment, avoid filling prescriptions and rely on episodic care. Though I agree with Linda Goettina that health insurance by no means ensures good care, being without feels like nothing less than a death sentence for many cancer survivors. Informed providers, increased research and educational efforts aimed at survivors will do little or nothing to help the uninsured cancer survivors.

Competing interests:   None declared

This paper is timely indeed – the consensus amongst health care providers, parents and survivors that gave rise to the report published by the National Cancer Board1 attests to that. The long term outcomes in cancer survivors reflect an interplay of the type and extent of treatment delivered, hereditable characteristics that may modify the impact of that treatment, and environmental exposures and modifiers, both chemical and physical. There is a panoply of potential adverse outcomes, and a moderately wide range of possibly effective pre- emptive interventions. While the size of the benefit from such interventions has not yet been accurately determined in this population, it is reasonable to extrapolate the benefits from other, less vulnerable populations. It is unreasonable to expect primary health care practitioners, or indeed many oncology practitioners to be knowledgeable about the specific risks for any particular survivor, given the variability bestowed by specific treatment, time since treatment and other variables. The development of Aftertreatment programs at cancer centers may address this issue for a proportion of survivors geographically close to the center, but for a large proportion, care will devolve on primary health care practitioners,which is precisely why the transfer of information and appropriate education is key to the provision of health care to this population. They are probably their own best advocates, if armed with the necessary information about their treatment. A significant proportion of subjects in this cohort were limited in knowledge of treatment they had undergone2 . It is clearly the obligation of the treating institution to provide such information to the survivor. The corollary of that is the obligation to provide education and resources to the healthcare providers. The greater likelihood that high risk patients report all types of clinical visits is re-assuring, but may reflect only clinically driven need to seek health care. The progressive drop off in cancer related health visits and general physical examination likely reflects a failure to convey the delayed onset of many of the impacts of survivorship to the survivors. It may also, however, reflect at least two other issues- systemic barriers to access, as reflected by the elevated likelihood of lack of visits in the small proportion lacking health insurance in this sample (a proportion lower than the national average), and a failure of health care providers to meet the needs of survivors. Table 4 suggests that even for the highest risk group, CNS tumor survivors, there is a progressive drop off in health care visits with advancing years which may reflect exactly the difficulty health care providers encounter in meeting the needs for inclusive, holistic care. Of considerable interest is the fact that 74% of this cohort identified themselves as concerned to very concerned for their future health, while only 42% reported a cancer related visit and 20% a cancer center visit – we need to do better than this. References: 1. Childhood Cancer Survivorship: Improving Care and Quality of Life. Hewitt M, Weiner S, and Simone J, eds. National Academies Press Washington D.C. 2003 2. Kadan-Lottick NS, Robison LL, Gurney JG, et al: Childhood cancer survivors' knowledge about their past diagnosis and treatment: Childhood Cancer Survivor Study. JAMA 2002: 287:1832- 1839.

Competing interests: None declared

I am a 47-year survivor of Wilms' tumor, treated at age 11 months in 1956, and a member of the Long-Term Survivors (LTS) online discussion list mentioned by Linda Goettina.

Dr. Kevin Oeffinger and his colleagues study of and subsequent article on "Health Care of Young Adults of Childhood Cancer" is a huge step forward dealing with issues for childhood cancer survivors and it brings these concerns to the forefront.

For many years I believed my cancer history was far behind me. I found out differently, as I aged, that an increasing number of abnormal symptoms developed. I believe if years ago I had been given some kind of protocol for long-term health care to follow, I might currently be in a better state of health. Therefore, I suggest that an individualized protocol be developed for childhood cancer survivors to have for safe- keeping for their long-term health care. It should be given to their parents when they are dismissed from active treatment by their oncologists. This protocol should also include their cancer diagnosis and date of treatments, and the types and amounts of treatment received (radiation and/or chemotherapy, surgeries, etc.). There are childhood cancer survivors today who no longer have parents or attending physicians living who can provide these answers. Some don't know what kind of cancer they had.

The medical community must be made aware that there can be late- effects for childhood cancer survivors. During many of my ER visits, I have been asked by the medical staff, "What do you mean by late-effects? I've never heard that term before." The Children's Oncology Group's "Childhood Cancer Survivor Long-Term Follow-Up Guidelines" should be readily available to all PCP's and ER physicians. This would provide them with a basic guideline to determine the best course of tests, treatments or referrals when presented with a long-term cancer survivor.

Unlike many LTS, I have a PCP who addresses my concerns. If she can't help me she will find someone who possibly can. I also finally have access to oncologists at the children's hospital where I was treated in 1956. They help me every three years with my disability reviews and document my late-effects, the only way I can prove eligibility for benefits. We need physicians who are aware of late-effects to help us with disability issues and many other concerns.

Competing interests:   None declared

I am a 50-year survivor of Wilms’ tumor and a member of the long-term survivors (LTS) internet discussion group mentioned by Linda Goettina (“Further Thoughts on Follow-up Care”). List members have often discussed the intense silence in the medical community on the long-term health care of all long-term cancer survivors. "Health Care of Young Adult Survivors of Childhood Cancer" is a beginning, and LTS welcome every effort made on their behalf and that of future survivors.

It is reassuring that Dr. Oeffinger and his research team concluded that “it is critical that cancer centers and primary care physicians develop methods to communicate…” This current lack of communication -- and an overall lack of knowledge and understanding of the extent of LTS problems in the medical community as a whole – leave an increasing number of survivors to suffer in ignorance, frustration and isolation. Internet discussion groups are rapidly filling the void, becoming a haven of information, understanding and community.

While the limitations of this study are indicated, it reveals for the first time what strictly clinical studies have not. I am writing to suggest another such study, one on the health care of older adult survivors of childhood cancers. While continuous medical records would be impossible, its findings would increase the knowledge of late effects experienced over 30, 40 and even 50 years. Opening communications with primary care physicians (PCP’s) and alerting them to watch for a cancer diagnosis in routine medical histories would unearth, I am sure, a surprising number of cases to study. The burgeoning number of personal web sites by cancer patients and survivors offers another source. For example, through my own personal web site alone (www.wilmstumor.us), I have been contacted by 45 survivors diagnosed prior to 1980, 9 of them prior to 1960, with the oldest diagnosis in 1947.

Such a study -- combined with archived medical records and an inquiry into the present medical condition of these survivors and how they have handled their late effects -- would greatly increase the overall understanding of the long term effects of cancer treatments and add to the chronicles of medical history. It would be expeditious to pursue this study as soon as possible, as most of the survivors from the early days of modern cancer treatment have died, and few of us remain.

Competing interests:   None declared

Oeffinger et al.(1) have quantified the familiar difficulties faced by childhood cancer survivors entering the adult health system. Even in this cohort treated at major cancer centers and motivated to participate in a demanding study, many are not adequately followed for their unique vulnerabilities. Examples of high-risk patients almost certainly not monitored for potentially life threatening late-effects are alarming given late mortality in this population.(2) The authors note the potential for patient report to introduce a pessimistic bias, as patients may receive medical monitoring they are not aware of. But the data may give an optimistic view of survivors’ follow-up care, since oncology visits may not universally include the risk-based screening advocated by the authors.

Difficulties in providing appropriate care to graduates of pediatric specialty care have been recognized(3), but cancer survivors face particular challenges. Unlike arthritis or diabetes patients, cancer survivors are not typically chronically ill, and don’t come to primary care with treatment histories and personal awareness highlighting their health needs. Rather, they are at risk for a variety of complications many years after treatment. It may be unclear what follow-up is optimal and what professionals should oversee this aftercare. In this context, the Oeffinger et al. results are not surprising. Systemic obstacles may include age limits in some pediatric cancer clinics, patient and provider mobility, insurance carrier changes, and limited coverage for oncology visits without a cancer diagnosis. Reported barriers to care include ethnicity, insurance and gender, though survivor-specific factors are likely involved. Expectations for primary care may be particularly important, as survivors may have trouble adjusting to care that is less frequent and less intensive than cancer treatment.

Knowledge of late-effects and effective screening are critical to insuring appropriate survivor follow-up, and the recommendation to develop technology to bring this information to patients and providers is precisely what is needed. But knowledge alone is often ineffective in promoting behavior change. Studies of survivor health beliefs and risk perceptions may help explain variability in follow-up care in this population. Survivors are faced with the difficult task of viewing themselves as “cured,” but still vulnerable to medical complications. Understanding how survivors can integrate these views of themselves will be important in promoting adherence to post-treatment care. Interventions to promote appropriate follow-up will need to begin very early in the off- treatment period so that survivors become informed and socialized in a view of survivorship that includes regular medical contact. 1. Oeffinger KC, Mertens AC, Hudson MM, Gurney JG, Casillas J, et al. Health care of young adult survivors of childhood cancer: a report from the childhood cancer survivor study. Annals of Family Medicine. 2004;2:61- 70. 2. Hudson MM, Mertens AC, Yasui Y, et al. Health status of adult long-term survivors of childhood cancer: a report from the Childhood Cancer Survivor Study. JAMA Sep 24 2003;290(12):1583-1592. 3. Goodman DM, Mendez E, Throop C, Ogata ES. Adult survivors of pediatric illness: the impact on pediatric hospitals. Pediatrics. 2002;110(3):583- 9. 2002.

Competing interests:   None declared

Oeffinger and colleagues provide a cogent and empirically based rationale for long-term health care and surveillance for survivors of childhood cancer. They emphasize the need to (1) educate survivors about long-term effects and health risks related to treatment, (2) enhance the transition of care to primary care providers, and (3) foster on-going communication between childhood cancer survivors, the cancer centers where they were treated, and primary care physicians. They also point out that factors typically associated with lack of health care in the general population -- lack of health insurance, ethnic minority status, and male gender -- were associated with limited nonspecific medical contact within a large cohort of childhood cancer survivors diagnosed and treated between 1970-1984.

A major finding of this report is that lacking health insurance increased the likelihood of long-term survivors not receiving risk-based health care. From the Childhood Cancer Survivor Study (CCSS) data collected 1994-1996, 20% of survivors age 18-24 and 15% of survivors age 25-34 did not possess health insurance (1). These rates are lower than the 29% and 22%, respectively, found in the US population for same-age adults in 1996 (2). With CCSS cohort participants being more likely to possess health insurance than same-age peers in the general population, the rates of health care utilization reported by Oeffinger and colleagues may be overestimates of actual follow-up care in the childhood cancer survivor population. Worse yet, US Census data indicate that the ranks of the uninsured have increased since 1996 with young adults between the ages of 18-34 being more likely than any other age group to be uninsured. In 2002, 30% of young people between the ages of 18-24 and 25% of persons between the ages of 25-34 lacked health insurance (3). Rates of being uninsured also were disproportionately high for ethnic minority groups and persons of low socioeconomic status in spite of the existence of Medicaid (3).

The ability to access adequate and appropriate long-term follow-up care for past, present and future cohorts of long-term cancer survivors may be adversely affected by inadequacies and inequalities in the US health care system. To better assure the health of our patients, families and nation, we need to act beyond the confines of clinical practice and advocate for an equitable and financially viable health care system that will be responsive to the health care needs of all persons living in the United States.

(1) Personal communication, Norman Turk, M.S., Statistician for the Childhood Cancer Survivor Study, UCLA School of Medicine, Department of Pediatrics (2) Bennefield, Robert L. Health Insurance Coverage – 1996, Current Population Reports, (September 1997), US Census (see http://www.census.gov/prod/3/97pubs/P60-199.PDF) (3) Mills, Robert J. and Bhandari, Shailesh. Health Insurance Coverage in the US: 2002. Current Population Reports, (September 2003), US Census (see http://www.census.gov/prod/2003pubs).

Competing interests:   None declared

Dr. Mahoney writes, “It is likely that a portion of those survivors not reporting any medical care lack health insurance since respondents were typically in their 20’s or 30’s and treatments may have interrupted educational/vocational training. Oeffinger et al.(5) did not provide comparisons of levels of medical contact from the general population, nor did they report whether these survivors were more or less likely to report having health insurance.” Dr. Mahoney raises a valid and important question. Perhaps the Long Term discussion list can shed some further light on the question of insurance. Since the membership is international, many members come from countries where universal access to health care is the norm. However, these members are just as likely to report the same difficulties in obtaining reasonable medical care and interventions as their American counterparts. It does not seem in these instances that lack of insurance or availability to health care is the limiting factor in seeking follow-up care.

Most of the members of the LTS discussion group are acutely aware of their need for continuing health insurance. Questions of coverage can spark long discussions. At least one aspect of the insurance question for survivors results in a certain deformation of life choices. One of the unspoken but quite common problems for survivors is the need to maintain health insurance at any cost. That means survivors often choose to stay in marriages and work situations that are ultimately unsatisfying and unhappy because they are afraid to lose their health insurance coverage. I can cite more than one marriage that continues despite real unhappiness because the spouse in the marriage provides the much need health care benefits. Survivors are not unaware of their need for good health care benefits and sometimes make real sacrifices to maintain benefits. But health insurance in and of itself is not a guarantee for good follow-up care.

Finally the last problem survivors with insurance face is the ongoing battle with HMO organizations that frequently deny referrals to clinics and physicians who specialize in the treatment of survivors. We have had more than one survivor who has had to do battle with their insurance provider in order to seek much needed care with someone knowledgeable about late effects. And I know of many survivors who have paid out of pocket to see those few physicians who understand the medical challenges we face.

Competing interests:   None declared

The report by Oeffinger and colleagues provides us with important and rather sobering data on the health care practices of adult survivors of childhood cancer. The results of this large cohort study, while not entirely unexpected, underscore the magnitude of the problem: How do we provide optimum health care that is both informed and accessible to this vulnerable population? Of great concern is the fact that the health problems that surivors face tend to increase as they age, at a time ,paradoxically, when they are less likely to have contact with specialists with expertise in cancer aftercare or "Late Effects".

The problem is compounded by the fact that these diseases occur rather infrequently and that this relatively small population of survivors is spread across the country. As the authors point out, primary care clinicians will likely remain the major source of medical care for childhood cancer survivors. Thus, resolution of this health care "crisis" is dependent upon collaboration and communication between the pediatric oncology and primary care communities. Moreover, solutions are likely to require strategies that are novel and innovative. From my perspective as a clinician caring for a large population of pediatric and adolescent cancer survivors, this is one of the biggest and most difficult challenges for the future.

Competing interests:   None declared

The latest report from the Childhood Cancer Survivor Study written by Oeffinger et al indicates that about 83%, 49% and 31% of survivors aged 18 -19 years reported a general physical examination, a cancer related visit to a physician’s office or a visit to a cancer centre, respectively. In contrast, the percentages of survivors aged 35-48 years reporting these types of visits declined to 67%, 38% and 17%, respectively. This raises concerns as the risk of many of the most serious late effects of cancer treatment, for example solid second primary cancers, increases with age and interval from diagnosis. Oeffinger et al noted that only 52% of survivors who had received at least 300 mg/m2 of anthracyclines reported a cancer-related visit to a physician’s office. Moreover, nearly 50% of Hodgkin’s disease survivors reported no such care.

In a research paper to appear in Pediatric Blood and Cancer in February 2004 we report some preliminary findings from the primary care physicians approached as part of the British Childhood Cancer Survivor Study*. We report that 35% of 10979 survivors representative of the entire study population were still on regular long-term hospital follow-up in relation to their childhood cancer. However, there was considerable variation with age: 64% of the 1319 survivors aged 16-19 years were still on such hospital follow-up, but only 12% of the 1984 survivors aged at least 40 years were still on such follow-up.

Oeffinger et al identify four key obstacles contributing to a decline in clinical follow-up with age and time elapsed since treatment: they relate to provision of information and its effective communication between hospital clinicians, primary care physicians and survivors.

Oeffinger et al also observe that the primary care physicians provide health care for most of the growing population of survivors of childhood cancer. The preliminary data from the British Childhood Cancer Survivor Study* indicate that 65% are de facto in the care of primary care physicians. We agree entirely with Oeffinger et al that the provision of information and its effective communication is the key and needs to be improved between hospital clinicians, primary care physicians and survivors. In our forthcoming paper in Pediatric Blood and Cancer we discuss the merits of the use of survivor-held records summarising diagnosis, treatment received and potential late effects. Such a summary could be produced at discharge from hospital-based follow-up and copies provided to both the survivor and their current primary care physician. Such patient held records would seem to have considerable potential benefit with little risk of doing harm.

Finally, we consider that work is essential to provide evidence-based guidelines on clinical follow-up.

*The British Childhood Cancer Survivor Study includes a postal questionnaire survey of 14500 survivors of childhood cancer who are aged at least 16 years and who were diagnosed between 1940 and 1991 inclusive in Britain. The postal questionnaire is sent via the primary care physicians who provide a limited amount of information concerning the survivor.

Mike Hawkins and Aliki Taylor

Centre for Childhood Cancer Survivor Studies Department of Public Health and Epidemiology The University of Birmingham BIRMINGHAM B15 2TT UK

E-mail: M.M.Hawkins@bham.ac.uk Tel: 0121 414 7924 Fax: 0121 414 7923

Competing interests:   None declared

The authors determined the healthcare utilization reported by 9,434 young adult survivors of childhood cancer, and examined factors associated with limited medical care. Outpatient medical care within the last two years was defined as one of the following four: i) general contact with the health care system, general physical examination, cancer-related medical visit, and medical visit at a cancer center. Factors associated with self-reported limited medical care included absence of health insurance, male sex, lack of concern for future health, and age greater than 30 years in comparison with that between 18 and 29 years. Increasing age and time from diagnosis, as well as exposure to therapy associated with an increased risk of adverse events such as congestive heart failure and secondary breast cancer were associated with a lower probability of healthcare utilization. Risk of most adverse events increases with increasing follow-up among patients who are exposed to therapies associated with these late effects. As the authors point out, survivors can benefit from early diagnosis and intervention or preventive care targeted at reducing risk for late effects. Although certain sociodemographic risk factors have been identified as associated with decreased healthcare utilization in this population, factors not examined include the presence of complications, disease status of the patients, patient, healthcare provider education and awareness of the patients and the healthcare providers of the potential for development of late effects and hence the need for life-long follow-up. All of these factors could very well determine who accessed medical care in this cohort. None-the- less, this study has done an excellent job in describing the pattern of healthcare utilization among the childhood cancer survivor population, and brings up an important issue of increasing the communication between the cancer centers and the primary health care physicians in order to optimize the life-long care of this growing population of childhood cancer survivors. Towards this end, the Children’s Oncology Group has developed Long-term Follow-up Guidelines that are available at www.childrensoncologygroup.org (click on “Long-Term Follow-Up Guidelines”). These guidelines serve as an excellent resource to the healthcare provider of the potential late effects associated with specific exposures, the population at risk for the development of these late effects, and the recommended screening tests, as well as their frequency. In addition, the Guidelines have a series of “Health Links” that serve as patient education material for the cancer survivors.

Competing interests:   None declared

In this article Dr. Kevin Oeffinger and his colleagues, reporting for the Childhood Cancer Survivor Study (CCSS), describe patterns of ongoing health care received by young adult survivors of childhood cancer. The CCSS cohort they studied is by far the largest in the world of individuals diagnosed with cancer during childhood and then followed for several decades. Fortunately, over three-quarters of children with cancer are now cured. However, the "quality" of these cures is suboptimal. Therefore, ongoing medical follow-up * with attention given to the unique late effects results from these individuals' cancers and their treatments * is required. Many, if not most, of these patients are no longer followed by pediatric oncology specialists. After all, they are no longer in the pediatric age group and no longer carry a diagnosis of cancer. Transitioning their care to adult cancer specialists is also not the appropriate follow-up strategy. So, the primary care physician bears the brunt of responsibility to see and manage these patients. Becoming familiar with their unique medical problems is therefore critical. As Dr. Oeffinger and his colleagues show, a sizeable percentage of these young adult survivors fail to consult physicians at all, and most have not recently visited a cancer center. The investigators have identified a number of barriers associated with these gaps in ongoing care and suggest methods to deal with them. The next steps are to test and refine novel monitoring and intervention strategies for this increasing population of cancer survivors.

Competing interests:   None declared

Oeffinger K, Mertens A, Hudson M, et.al.: Heatlh Care of Young Adult Survivors of Childhood Cancer: A Report from the Childhood Cancer Survivor Study.

Commentary Wendy Hobbie

In the future, one of the great challenges for pediatric oncology will be to prepare the adult health care system for the specialized needs of childhood cancer survivors. Many survivors currently lead healthy lives, however, there is a subset of individuals who are at risk for late effects. Furthermore, all cancer survivors must have information on their disease, treatment, risks and health promotion to modify risk factors in an attempt to attenuate late effects.

Oeffinger et.al. clearly and concisely review interesting data regarding survivors’ interaction with health care. Specifically, the majority of childhood cancer survivors are not returning to cancer centers for specialized care and are therefore not receiving the education needed to promote early identification of late effects.

The authors point out that “to optimize risk-based care, it is critical that cancer centers and primary care physicians develop methods to communicate effectively and longitudinally”. These survivors need to be transition to the adult health care system, but if we lose the critical link to the treating cancer center, the results could be catastrophic. If adult health care providers don’t have a resource to obtain the information necessary to provide comprehensive care, then their ability to evaluate and develop a treatment plan will be limited. Conversely, if pediatric oncology loses to follow-up these young adults as they age, critical information will be lost in order to alter current therapies to reduce late effects for the next generation of survivors.

Oeffinger, et. al. identify roadblocks to fostering relationships between cancer centers and practices that include lack of information, and survivors representing a small percentage of their practices. However, there are 9.6 million individuals who are surviving cancer, so this lack of attention is unjustified.(NCI) A collective effort between adult oncology, pediatric oncology and the adult primary health care system is necessary to provide care to this at-risk group who are living well, but require specialized care as they age due to the increasing risk of late effects. Nurse practitioners may be one means of providing a critical link when transitioning care and information to the adult cancer center and adult primary health care system, while maintaining a link to pediatric oncology.

Reflecting on the words of Dr. D’Angio (1975)…”a parallel effort is required in oncology so that the children of today don’t become the chronically adults of tomorrow”. Now that these children are adults, we have a responsibility to assist them in living life to the fullest with minimal effects from their therapy.

NCI- http://cancercontrol.cancer.gov/ocs/prevalence/index.html

D'Angio G: Pediatric cancer in perspective: Cure is not enough. Cancer 35:867-870, 1975 ( supplement)

Competing interests:   None declared

"Health Care of Young Adult Survivors of Childhood Cancer: A Report from the Childhood Cancer Survivor Study" provides a wonderful look at a growing problem facing the American health care system.

As a young adult cancer survivor I have experienced first hand the barriers associated with receiving coordinated quality long-term survivorship care.

In my role as Director of Survivorship at the Lance Armstrong Foundation and in founding the Ulman Cancer Fund for Young Adults, I am reminded daily of the problems that long-term cancer survivors face when trying to obtain medical care over the course of their survivorship.

Survivors often have difficulty finding a qualified physician who has cared for other survivors and they also struggle mightily to gain access to their records so that their new family physician can review and determine their follow-up care. This lack of coordination between the treating institution and the community physician must improve if long-term survivors will ultimately obtain the care they need.

Additionally, survivors and their families tend to have a warm, open relationship with their oncologist for many years following treatment, but that same dynamic doesn't always exist with their new primary care physician. This lack of a significant relationship between patient and physician often causes survivors to downplay some potential symptoms and or health concerns that may arise for fear that the issues at hand are not worthy of a visit to the doctor, or even a phone call.

On a personal note, when I feel run down or am experiencing symptoms that could be related to my cancer survivorship I email my oncologist, who is more than 1000 miles away, to first determine whether my concern is legitimate. It would be much easier to simply call my local physician but there is a barrier there that I have not yet been able to hurdle.

This article outlines the issues at hand and I am most certain it will allow the process of opening the communication lines between family physicians and institutions that treat children and adolescents with cancer to begin.

A special thank you to the authors and those involved with the Childhood Cancer Survivor Study for all that they are doing to bring heightened awareness to these issues.

Competing interests:   None declared

Survivors of childhood cancers are at increased risk of developing a variety of late medical complications as a result of prior cancer therapy (1-3) and health status is also negatively impacted.(4) The paper by Oeffinger & colleagues indicates that although 87% of childhood cancer survivors reported some contact with the medical care system during the preceding two years, reports of a general physical examination, a cancer- related visit, and a medical visit at a cancer center were more modest.(5) These data suggest that those patients most in need of surveillance and medical follow-up report less use of health care; questions on the scope of services obtained by those reporting use remain unanswered.

It is likely that a portion of those survivors not reporting any medical care lack health insurance since respondents were typically in their 20’s or 30’s and treatments may have interrupted educational/vocational training. Oeffinger et al.(5) did not provide comparisons of levels of medical contact from the general population, nor did they report whether these survivors were more or less likely to report having health insurance. Moreover, further examination of psychosocial issues would be interesting.

The heterogeneity among childhood cancer survivors in diagnoses, era of treatment, types of therapy, and interval since diagnosis provides an opportunity for emphasizing communications between primary care clinicians and oncologists, as well as other physicians specializing in late effects. These interactions can be useful in detailing prior treatments and organizing surveillance plans tailored to individual patients, as well as generating enhanced contributions on this topic to the primary care literature.(5) Childhood cancer survivors may demonstrate deficits in knowledge regarding their diagnosis and prior treatment;(6) accordingly, access to medical records is imperative.

With greater recognition of these late effects, oncologists have instituted a variety of refinements designed to minimize long term complications including changes in doses and schedules for radiotherapy, enhanced monitoring during treatment with modification to chemotherapy dose, exploration for less toxic agents, and reduced use of prophylactic cranial radiation therapy.(3)

Because published data on childhood cancer survivors are generally limited to cross-sectional studies and cohort studies of modest size, it has not been possible to develop evidence based management recommendations.(1) Nonetheless, recommendations for surveillance are based upon the best evidence presently available. Clinicians are encouraged to become familiar with the scope of variety of late effects which are possible (see http://www.cancer.umn.edu/ltfu). With more than 270,000 survivors of childhood and adolescent cancer in the United States, most of us will encounter such patients in our practices.

References:

1. Oeffinger KC. Childhood cancer survivors and primary care physicians. J Fam Pract. Aug 2000;49(8):689-690.

2. Sklar CA. Overview of the effects of cancer therapies: The nature, scale and breadth of the problem. Acta Paediatr Suppl. Dec 1999;88(433):1- 4.

3. Marina N. Long-term survivors of childhood cancer. The medical consequences of cure. Pediatr Clin North Am. Aug 1997;44(4):1021-1042.

4. Hudson MM, Mertens AC, Yasui Y, et al. Health status of adult long-term survivors of childhood cancer: a report from the Childhood Cancer Survivor Study. JAMA Sep 24 2003;290(12):1583-1592.

5. Oeffinger KC, Mertens AC, Hudson MM, Gurney JG, Casillas J, et al. Health care of young adult survivors of childhood cancer: a report from the childhood cancer survivor study. Annals of Family Medicine. 2004;2:61- 70.

6. Kadan-Lottick N, Robison LL, Gurney JG, et al. Childhood cancer survivors' knowledge about their past diagnosis and treatment: Childhood Cancer Survivor Study. JAMA.287:1832-1839.

Competing interests:   None declared