Health care for childhood cancer survivors

Insights and perspectives from a Delphi panel of young adult survivors of childhood cancer

Authors

  • Brad J. Zebrack Ph.D., M.S.W.,

    Corresponding author
    1. University of Southern California School of Social Work, Los Angeles, California
    • University of California School of Social Work, 669 W. 34th St., Los Angeles, CA 90089-0411
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    • The authors are indebted to the twenty participants of this panel. Because anonymity for the survivors was desired, their names are not listed.

    • Fax: (213) 821-2088

  • Debra A. Eshelman R.N., M.S.N., C.P.N.P.,

    1. Department of Hematology/Oncology, Children's Medical Center of Dallas, Dallas, Texas
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    • The authors are indebted to the twenty participants of this panel. Because anonymity for the survivors was desired, their names are not listed.

  • Melissa M. Hudson M.D.,

    1. Department of Hematology Oncology, St. Jude Children's Research Hospital, Memphis, Tennessee
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    • The authors are indebted to the twenty participants of this panel. Because anonymity for the survivors was desired, their names are not listed.

  • Ann C. Mertens Ph.D.,

    1. Division of Pediatric Epidemiology and Clinical Research, University of Minnesota, Minneapolis, Minnesota
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    • The authors are indebted to the twenty participants of this panel. Because anonymity for the survivors was desired, their names are not listed.

  • Kelly L. Cotter J.D.,

    1. National Childhood Cancer Foundation, Bethesda, Maryland
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    • The authors are indebted to the twenty participants of this panel. Because anonymity for the survivors was desired, their names are not listed.

  • Barbara M. Foster Ph.D.,

    1. Department of Family Medicine, University of Texas Southwestern Medical Center, Dallas, Texas
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    • The authors are indebted to the twenty participants of this panel. Because anonymity for the survivors was desired, their names are not listed.

  • Lauren Loftis B.S.,

    1. Department of Family Medicine, University of Texas Southwestern Medical Center, Dallas, Texas
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    • The authors are indebted to the twenty participants of this panel. Because anonymity for the survivors was desired, their names are not listed.

  • Margaret Sozio B.S.,

    1. Department of Family Medicine, University of Texas Southwestern Medical Center, Dallas, Texas
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    • The authors are indebted to the twenty participants of this panel. Because anonymity for the survivors was desired, their names are not listed.

  • Kevin C. Oeffinger M.D.

    1. Department of Family Medicine, University of Texas Southwestern Medical Center, Dallas, Texas
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    • The authors are indebted to the twenty participants of this panel. Because anonymity for the survivors was desired, their names are not listed.

    • Dr. Oeffinger is a Robert Wood Johnson Foundation Generalist Physician Faculty Scholar.


Abstract

BACKGROUND

Most children diagnosed with cancer are surviving into adulthood but are not receiving adequate or appropriate follow-up health care. However, to the authors' knowledge, there is little literature published to date exploring potential barriers to long-term risk-based follow-up care for young adult survivors of childhood cancer.

METHODS

In the current study, using a modified Delphi technique, young adult cancer survivors identified barriers to utilizing appropriate follow-up care and offered suggestions for ways to enhance health care in this young adult population.

RESULTS

Major barriers to health care were found to be a lack of knowledge on the part of both physicians and survivors regarding long-term health issues related to cancer. Suggestions to enhance care included self-advocacy training for survivors and advanced training for primary care physicians who may treat childhood cancer survivors as they transition into adulthood.

CONCLUSIONS

The results of the current study are consistent with reports that young adult survivors of childhood cancer need or desire information regarding their medical histories, psychosocial support, and social advocacy. Cancer 2004;100:843–50. © 2004 American Cancer Society.

A recent report by the National Cancer Policy Board (established through the Institute of Medicine) indicates that survivors of childhood cancer are in need of long-term follow-up care and monitoring for late physiologic and psychosocial effects.1 This need is documented in long-term survivor studies that detail sequelae of treatment that place significant subsets of the population at risk for health problems later in life.2–7 For example, a recent study reported that nearly one in every two adult survivors of childhood cancer has at least one moderate to extreme adverse health outcome.8

Experts in the care of childhood cancer survivors suggest that lifelong follow-up care be founded on an empirically based and systematic plan for screening, surveillance, and prevention, and that this plan incorporate a survivor's risks based on the previous cancer and cancer therapy, genetic predispositions, lifestyle behaviors, and comorbid health conditions.9–12 However, to our knowledge there remains a lack of consensus regarding how and where to best implement a plan for follow-up as these young people transition from pediatric to adult models of health care.

Faced with a complex system of health care delivery and financing, young people with special health care needs are finding it difficult to locate and access providers who will become an essential part of their recovery and long-term survival.13 Due to myriad and often unknown circumstances, most young adult survivors of childhood cancer are not receiving adequate or appropriate health care.14

To our knowledge to date, there is little literature exploring potential barriers to long-term risk-based follow-up care for young adult survivors of childhood cancer. Furthermore, potential methods to enhance care for this relatively small (relative to survivors of adult cancers), diverse (in medical and demographic terms), geographically mobile, and growing population have yet to be systematically identified, implemented, or evaluated. There also remains a lack of consensus with regard to the most efficient means, in terms of financial and opportunity costs to both institutions and survivors, for providing that care.

To understand how to improve on the current status of care of cancer survivors, it is important to elicit perspectives from the three primary groups involved in the process: survivors, health care professionals, and policy experts. The first of these, the perspective of the survivor, is noticeably absent from the literature, yet their unique perspective as “health care consumers” is critical in the process of improving care. Thus, the purpose of the current study was threefold: 1) to identify barriers/obstacles to obtaining appropriate follow-up care; 2) to elicit suggestions for components and models of care having the greatest potential for success; and 3) to assess potential ways to enhance health care for the survivor population, all from the perspective of young adult survivors of childhood cancer. The Delphi method was used to meet these goals by assembling a panel of survivors with a breadth and depth of survivorship experience on a national, state, or local level. Our intent was to elicit participation from individuals who could articulate and reflect key issues and struggles in the health care of survivors of childhood cancer.

MATERIALS AND METHODS

The Delphi Process

Briefly, the Delphi method is a qualitative research tool that provides a means for a panel of experts from a variety of settings to formulate a list of ideas, followed by the development of group consensus concerning the relative importance of those ideas.15–18 An expert is defined as one who has demonstrated knowledge regarding a particular subject and an ability to articulate a broad perspective on an issue. Panel members participate by responding anonymously to a questionnaire comprised of several iterations, or rounds. Questions included in each successive round depend on the responses from the previous round. Consensus is eventually attained by rank ordering responses and quantifying attributions of degree of importance.

Participants

The goal of assembling the Delphi panel for this study was to include individuals representing a diversity of survivorship experiences and backgrounds. To capture the desired breadth of ideas and concepts from such a group, it was essential that the participants be able to articulate these experiences as well as represent the views of others. Potential panelists were selected based on a documented history of participating in efforts regarding cancer survivorship or advocacy at a national, state, or local level (paid or volunteer basis). Importantly, it was believed that this level of experience was required to reflect the views of a wide constituency of survivors, including survivors who are actively involved in risk-based health care as well as those who have limited access or interest in such care. As with any “expert” panel, their views represent their own experiences and biases as well as those of the many survivors with whom they have interfaced.

Twenty-one such individuals were invited to participate on the Delphi panel and 20 agreed to participate. Of these, 19 panelists were survivors of malignancies diagnosed during childhood, adolescence, or young adulthood (before the age of 30 years), and 1 was the mother of a deceased cancer patient. The demographic characteristics of the panel are summarized in Table 1.

Table 1. Demographics of Survivor Panelistsa
 Frequency
  • CNS: central nervous system.

  • a

    Does not include the mother of a deceased young adult survivor who had Hodgkin disease.

Gender 
Female11
Male8
Diagnosis 
Hodgkin disease6
Non-Hodgkin lymphoma5
Soft tissue sarcoma2
CNS tumor2
Bone tumor1
Leukemia2
Wilm tumor1
Geographic representation 
Texas5
Maryland4
California2
New York2
Tennessee2
Arkansas1
Connecticut1
Illinois1
Pennsylvania1
Rural7
Suburban7
Urban5
Median age (yrs) (range) 38 (21–51)
Median age at diagnosis (yrs) (range) 13.9 (1–27)

The study was approved by the institutional review board of the University of Texas Southwestern Medical Center at Dallas. All participants were blinded to each other's identities and the research team was blinded to the identities of each panelist. Between March and August of 2002, the panelists completed a series of three questionnaires. All 20 participants completed and returned the Round One questionnaire. Three participants passively dropped out of the panel after the first round and did not complete the final two rounds. The remaining 17 participants completed and returned questionnaires from the second and third rounds.

The data were collected, analyzed, and interpreted by a multidisciplinary team comprised of a social scientist (B.J.Z.), three clinicians (K.C.O., M.M.H., and D.A.E.), a statistician with expertise in qualitative methodology and the Delphi method (B.M.F.), an epidemiologist (A.C.M.), a lawyer with experience in survivorship advocacy-related work (K.C.), a second-year medical student (M.S.), and an undergraduate student (L.L.). The social scientist, the three clinicians, the epidemiologist, and the lawyer each have many years of experience working with cancer survivors and investigating their health care needs. The three clinicians are from different backgrounds: a family physician who directs a program for young adult cancer survivors; a pediatric oncologist who directs a program for survivors of pediatric cancers; and a pediatric oncology nurse practitioner who coordinates a program that provides care for childhood, adolescent, and young adult survivors. This breadth and depth of backgrounds was important when analyzing data and interpreting the findings.

RESULTS

Round One

The goal of the first round was to generate ideas for the derivation of items to be evaluated by panelists in future rounds. Panelists were asked to: 1) list potential barriers or obstacles to optimal health care for adult survivors of childhood cancer; 2) describe or diagram an idea or vision of what optimal long-term health care of adult survivors of childhood cancer should be; and 3) describe possible ways to enhance the health care of this group. One page was provided for responses to each question, with additional pages provided at the end of the questionnaire. Members of the research team collated, sorted, and edited responses to achieve consistent wording of items expressing similar thoughts or ideas.

After editing responses from the first round, a total of 44 items were created for Question 1 –(Potential Barriers to Health Care). These items were grouped into 4 categories: Survivor-related Barriers (14 items), Psychological Barriers (6 items), Provider-related Barriers (15 items), and Insurance or System-related Barriers (9 items). From the responses to Question 2– (Models of Care), 27 responses (and 1 Diagram) were categorized into Components of Care (13 items), Types of Health Care Providers (10 items), and Settings for Delivery of Health Care (4 items). Twenty-six responses listed for Question 3– (Ways to Enhance Care) were organized into Individual Survivor (4 items), Health Care Provider (10 items), and Health Care System/National Policy (12 items) categories.

Round 2

The goal of the second round was to identify the dominant and most highly endorsed ideas and themes for each of the three central questions. A 13-page questionnaire was developed for the second round that was comprised of all responses listed in the first round and grouped into the categories described earlier for each of the 3 central questions. Panelists were asked to: 1) score the importance of each item on a 7-point Likert scale with anchors ranging from “not important” to “critically important”; 2) rank order the five most important items within each category; and 3) add further items to the list, if necessary. Responses from this round again were sorted and edited for redundancy. The mean and median importance scores for each item were calculated. The number of times an item was ranked first, second, third, fourth, or fifth and the number of times it was not ranked was assessed. The items and their average ratings of importance for each of the three questions are summarized in Tables 2–4.

Table 2. Mean Scores of Importance for Barriers and Obstacles to Health Care for Young Adult Cancer Survivorsa
CategoryItemsMean scoreb
  • HMOs: health maintenance organization.

  • a

    Because of the large volume of items in response to this question, only items with mean score ≥ 5 are included in the table.

  • b

    On a scale of 1–7, with 1 = not important at all and 7 = critically important.

Survivor-relatedSurvivors lack knowledge about late effects and are unaware of their risks6.41
 Survivors do not know the details of their disease and treatments5.47
 Lack of personal finances for health care5.35
 Survivors are concerned about losing (or not getting) insurance if a medical problem is discovered5.12
PsychologicAnxiety or fear of being diagnosed with cancer again6.47
 Survivor's desire to “put cancer in the past” to just “move on”5.94
 Anxiety or fear of being diagnosed with a chronic disease caused by cancer treatment5.94
Health provider-relatedLack of training/education of primary care physicians about health problems of survivors6.47
 Lack of training/education of specialists regarding health problems of survivors6.47
 Limited number of adult health care providers equipped to deal with childhood cancer survivors6.29
 Physicians often fail to differentiate health issues as related or unrelated to previous cancer5.81
 Lack of communication among specialists; need for a cooperative medical team5.71
 Lack of support for transition from pediatric to adult health care5.35
 Lack of importance placed on issues of survivorship by “Pediatric Oncology”5.25
 Lack of adult medical community's interest or time in caring for young adult survivors5.25
 Lack of consensus about what tests/evaluations need to be done and how often5.18
Health system-relatedLack of insurance6.59
 Restrictions of HMOs; HMOs that limit providers to members of their network6.44
 Lack of availability of follow-up programs6.18
 Lack of information about appropriate follow-up programs6.06
 Finding good, affordable care that provides for referrals to specialists5.71
 Difficult to access/locate medical records5.35
Table 3. Mean Scores of Importance for Components, Types of Health Care Providers, and Settings for Models of Optimum Health Care for Young Adult Cancer Survivors
CategoryItemsMean scorea
  • a

    On a scale of 1–7, with 1 = not important at all and 7 = critically important.

ComponentsFocus on educational activities regarding late effects and ways to lower a survivor's risk6.18
 Accessible medical record of the survivor that includes cancer and treatment information6.18
 Focus on screening for late effects6.12
 Ongoing communication between all of the health care providers6.12
 National web-based information center with information about late effects and links to resources6.00
 Inexpensive care5.71
 Uncomplicated and convenient access to specialty providers5.65
 Focus on wellness and prevention5.41
Types of health care providersPrimary care physician6.35
 Late effects “specialist”6.12
 Specialists5.76
 Psychologist4.65
 Nurse practitioner/nurse educator4.53
 Pediatric oncologist4.29
 Nutritionist/dietician4.29
 Social worker3.94
 Physical therapist3.94
 Alternative of complementary health care practitioner3.59
Settings for careA long-term follow-up clinic staffed by physician experienced with late effects and a nurse practitioner; based at a teaching hospital or cancer center but separate from the children's hospital/children's cancer center.6.06
 Single primary care physician (family physician, internist, gynecologist) in the community who would communicate with the cancer center/long-term follow-up program.5.53
 Multispecialty clinic/office that is away from the hospital and includes physicians from different specialties.5.53
 Long-term follow-up program staffed by a pediatric oncologist and nurse practitioner and based at a children's hospital or children's cancer center.4.24
Table 4. Mean Scores of Importance for Initiatives to Enhance Health Care for Young Adult Cancer Survivors
CategoryItems by rank orderMean scorea
  1. On a scale of 1–7, with 1 = not important at all and 7 = critically important.

Survivor-relatedEducate survivors about late effects6.65
 Educate survivors about how to be empowered, to advocate for themselves, to locate resources6.35
 Make sure survivors and families know about support groups and Internet forums and lists5.24
 Small, stable support groups are needed4.94
Health care provider-relatedEducate health care professionals about survivors and late effects6.59
 Provide a written medical synopsis to each survivor that includes a summary of all chemotherapy, radiation, operative and pathology reports and current medical problems and medications; help survivors maintain or rebuild their medical history6.47
 Develop and implement protocols for follow-up visits based on the previous cancer and treatment6.29
 Train physicians to discuss and address medical history of previous cancer5.76
 Provide honest assessment of likelihood for late effects5.71
 Teach physicians to listen and take their patients seriously5.65
 Encourage interdisciplinary cooperation in the medical community5.59
 Provide ready access to counseling/support groups5.29
 Create a board certified specialty for late effects5.06
 Provide holistic care, including nutrition4.53
Health care system or national policyMore research in late effects, both physical and psychologic emotional effects6.59
 Establish more long-term follow-up clinics6.18
 Develop system of follow-up in collaboration with insurers so that affordable health insurance coverage is available for long-term follow-up6.06
 Develop and maintain proper transition from pediatric to adult health care providers6.00
 Funding for people who have been denied insurance because of their histories, even though they work5.75
 Mail late effects findings to survivors with instructions of what they should do5.59
 National subsidy or insurance for survivors5.41
 Provide option for registering with a database that would follow survivors as they move5.29

Round 3

The goal of the third round was to gain consensus on the ranking of the key items for each question. After analysis and grouping of similar items from the second round, a 5-page questionnaire was mailed to panelists. Items included in the questionnaire for the third round were selected as a result of being the most highly endorsed items, in terms of aggregate rankings and average degree of importance, in the second round. In most categories, often-endorsed items from the second round were clearly distinct from less important items and were included in the questionnaire for the third round. To minimize bias, items that appeared important to some respondents but were ignored by others were included in the third round questionnaire. For the third round, respondents were asked to rank order 10 barriers to care and 9 potential initiatives to enhance the health care of survivors. The mean rank for each item was calculated. Table 5 summarizes the top five ranked barriers and the top five initiatives for enhancing care, as rated by respondents in the aggregate.

Table 5. Consensus Rank Order of Barriers to Care and Initiatives to Enhance Care for Young Adult Cancer Survivors
CategoryItems by rank order
Barriers1. Survivors lack knowledge about late effects and are unaware of their risks
 2. Lack of insurance
 3. Lack of training/education of primary care physicians about health problems of survivors
 4. (Tie) Lack of training/education of specialists regarding health problems of survivors
 4 (Tie) Limited number of adult health care providers equipped to deal with childhood cancer survivors
Initiatives to enhance care1. Education of health care professionals about survivors and late effects
 2. Education of survivors about late effects
 3. Provide a written medical synopsis to each survivor that includes a summary of all chemotherapy, radiation, operative and pathology reports and current medical problems and medications; help survivors maintain or rebuild their medical history
 4. More research in late effects, both physical and psychological and emotional effects
 5. Develop and implement protocols for follow-up visits based on the previous cancer and treatment

Also in the third round, panelists were asked to rank seven components of care, three preferred sources of care, and two settings of care based on notions of “best” and “most practical” care. The panelists' rankings of components and sources of care are summarized in Table 6. With regard to settings of care for adult survivors of childhood cancer, respondents elected as “best setting” a long-term follow-up clinic staffed by a physician experienced with late effects and a nurse practitioner, and based at a teaching hospital or cancer center but separate from the children's hospital/children's cancer center. In contrast, panelists suggested that a more “practical” model would involve a single primary care physician (family physician, internist, or gynecologist) in the community who would communicate with a cancer center or long-term follow-up program.

Table 6. Consensus Rank Order of Models of Care for Young Adult Cancer Survivors
CategoryItems
Components of optimum health care1. Focus on educational activities regarding late effects and ways to lower a survivor's risk
 2. Focus on screening for late effects
 3. Accessible medical record of the survivor that includes cancer and treatment information
 4. Uncomplicated and convenient access to specialty providers
 5. Ongoing communication between all of the health care providers
 6. Inexpensive care
 7. National web-based information center with information about late effects and links to resources
Preferred source of care1. Primary care physician knowledgeable and experienced in working with survivors
 2. Late effects “specialist”
 3. Specialists

DISCUSSION

Clinicians and researchers in the fields of pediatrics, medical oncology, and psychosocial and behavioral oncology concede that a large proportion of childhood cancer survivors are not receiving adequate or appropriate long-term health care. To our knowledge, explanations for why this may be have not been evaluated systematically. Thus, the current study represents an important contribution to understanding potential barriers to long-term care for young adult cancer survivors and suggests methods for overcoming these barriers, as seen from the perspective of educated, informed, and proactive survivor advocates. This perspective reported herein reflects the collective wisdom of a group of survivors who have learned to navigate a complex medical system in the U.S. The individuals in the panel have experienced, personally and by second-hand accounts, both positive and negative aspects of the health care afforded cancer survivors, and thus provide an insightful point of view of young adult consumers, a critically important perspective that is lacking in the current body of literature.

Overall, participants in our Delphi panels found barriers existing at both individual and broader systems levels. First, panelists reported a lack of critical information regarding their cancer and its treatment. Indeed, recent work indicates that a notable proportion of survivors lack information concerning their cancer diagnosis and treatment.7 A dominant theme throughout the current study was the importance of survivors being able to find and access information. Not possessing critical information regarding the type of cancer, chemotherapy exposure, radiation exposure, location and dosages, and the risks for late or delayed effects of therapy threatens survivors' future health. At the system level, the Delphi panelists suggested that their preferred sources of medical care–primary care physicians–lack appropriate knowledge and training regarding the health risks confronting childhood cancer survivors.

To overcome these obstacles, survivors emphasized the importance of possessing information concerning disease and long-term health issues. They also indicated the importance of being able to obtain guidelines for long-term follow-up care and access to providers qualified to provide this care. Panelists suggested the need for formal interventions that will 1) train and educate health care professionals regarding the risks of this population, 2) develop and implement protocols for follow-up visits based on the previous cancer and treatment, and 3) improve communication between childhood cancer-treating institutions and primary care physicians.

Regardless of where survivors may receive the “best” or “most practical” medical care and long-term follow-up, the notion of survivors being expert about their own cancer experience and having the ability to advocate on their own behalf was evident among panelists. This viewpoint is exemplified by one survivor's comment: “A very important component of each of these [long-term follow-up] programs should be the patient as self-advocate. My own care has improved since my primary care physician has included me in every decision and has started paying attention to my theories.”

Survivor panelists also expressed concerns regarding insurance and financial burdens as major system barriers. Recognizing the financial restrictions of our current health care system, several panelists suggested that survivors become advocates for themselves, seeing this as a viable alternative to making costly health care system changes. One panelist commented: “Most of the system changes described here are expensive and unlikely to happen in the near future. A less expensive alternative which wasn't mentioned is to provide education and tools for survivors that empower them to navigate systems and advocate for access and coverage of survivors.”

In evaluating the generalizability of the current study, potential biases and limitations of the Delphi method should be considered. First, there is a panel bias in that the views expressed were primarily those of the panelists. Second, the demographics of the panelists lacked racial/ethnic diversity. Third, the panel did not include survivors who practice avoidance or wish to forget their cancer experience. This is a common coping strategy that affects the health care utilization practices of survivors. That being said, the panel of survivors represents many years of experience in working on survivorship issues on local, state, or national levels. Their views reflect a breadth and depth of experience as lived through their own cancer and survivorship stories as well as through those of the countless survivors that they have met or worked with throughout the country. It is important to note that the study had a high level of completion by panelists, in comparison with other published Delphi panels.15–18 Finally, the empiric findings reported in the current study are not meant to be definitive conclusions with respect to the needs of young adult cancer survivors'. The aim of the Delphi method is to generate ideas and hypotheses for applications in broader and more representative samples of survivors, not to test them. Our intention in conducting this research was to provide a working framework that generates questions for further exploration and areas for potential intervention.

Conclusions

The results of the current study are consistent with reports that young adult survivors of childhood cancer need or desire information regarding their medical histories, psychosocial support, and social advocacy.19, 20 Achieving these objectives can come about by making resources available to health care professionals (e.g., clinical social workers) who are specifically trained to teach this population to advocate for itself. National organizations serving young adult cancer survivors, such as the Lance Armstrong Foundation, the Ulman Family Fund for Young Adults, Planet Cancer, Fertile Hope, and the Children's Oncology Camp Foundation/Young Adult Survivor Conference, have the potential to serve as productive and reliable sources of information, support, and advocacy training for this population. Furthermore, attention to creating and enhancing workable health care delivery and financing systems that engage and serve this population of survivors are necessary. These efforts must involve renewed attempts to legislate appropriate reimbursement, coordinate services, disseminate medical information, and determine evidence-based biopsychosocial protocols for the long-term follow-up of childhood cancer survivors who, among all cancer patients, represent the greatest potential for years of life-long productivity and social contribution.

Ancillary