A questionnaire based review of long-term follow-up programs for survivors of childhood cancer in Canada

Abstract

Background

Major advances have been made in the treatment of childhood cancer; however, survivors of childhood cancer are at increased risk for morbidity and mortality. There is little literature regarding available long-term follow-up programs for survivors of childhood cancer.

Procedure

In March 2007, 16 surveys were sent to pediatric hematology/oncology programs across Canada to determine what programs were available for survivors of childhood cancer, and the nature of such programs.

Results

Of 15 participating centers, 13 (87%) have multi-disciplinary programs for the long-term follow-up of pediatric cancer survivors. Research databases were documented in 9/15 (60%) of centers to document late effects. Dedicated programs for adult survivors of childhood cancer were established in 8/15 (53%) of centers. Access to subspecialty care for survivors was rated as quite good. Concerns were raised by many participants about patients being lost to follow-up. Respondents indicated that primary care physicians appear to be under-represented within dedicated long-term follow-up programs.

Conclusion

Long-term follow-up programs for survivors of childhood cancer are available in 87% of Canadian pediatric oncology centers. While programs reported good access to care for childhood survivors, many adult survivors of childhood cancer have more limited timely access to services and patients are often lost to follow-up. New models of care incorporating primary care physicians are necessary due to growing numbers of survivors. Pediatr Blood Cancer 2009;52:113–115. © 2008 Wiley-Liss, Inc.

INTRODUCTION

The field of pediatric oncology has seen tremendous improvements in long-term survival over the past 40 years 1. The overall cure rate for all types of childhood malignancies is now over 80%. However, with increasing numbers of survivors, there continues to be a growing understanding of the late effects of therapies, and it has been well described that there is an increased risk of morbidity and mortality for this population 1–4. As a result, a model of specialized care in long-term follow-up clinics dedicated to providing comprehensive, evidence-based health care and education has been adopted by some centers.

Such clinics have been established in increasing numbers over the years, and in 1997 a report indicated that 53% of institutions affiliated with the Children's Cancer Group and the Pediatric Oncology Group had a long-term follow-up clinic for survivors of childhood cancer 2. A recent report published in 2006, which surveyed 28 directors of well-established long-term follow-up clinics, indicated that even these leading programs in the field encounter many challenges in providing what they believe to be the standard of care for their patients 1. Such barriers included financial and manpower restraints, lack of commitment on the part of their institution and an increasing number of patients. Respondents also found that identifying primary care physicians to care for this complex and growing patient population a challenge.

In the United States, 1 in every 640 young adults is a survivor of childhood cancer, and there are 270,000 survivors of childhood cancer in total 5. Comparable numbers adjusted for Canada's population would be expected. Given the many challenges faced by even the most well established long-term follow-up programs, we believed that a survey of Canadian programs would provide important insight into the services provided across the country for this population which continues to outgrow its financial and personnel support 2, 6.

METHODS

A ten page questionnaire was developed and distributed to the heads of division at the 16 pediatric hematology/oncology centers across Canada in March 2007. These administrators were contacted by electronic mail, with an attached covering letter and copy of the questionnaire. It was asked that the questionnaire be completed by the person they deemed the most appropriate respondent in the division. Questionnaires were returned by electronic mail as well as by regular post over a 3-month period. All questionnaires were distributed in English; however, respondents were informed that a translated copy in French would be provided upon request. In some cases, participating centers were contacted several times (unless they declined to participate).

The questionnaire was divided into two sections. The first section included questions regarding programs for pediatric survivors of childhood cancer, with the second section addressing programs for adult survivors of childhood cancer. Questions in both sections focused on whether formal programs existed for survivors of childhood cancer, and what services were available. The organization, frequency and duration of follow-up as well as the criteria for inclusion in such programs were determined. Questions also pertained to the funding of long-term follow-up programs and access to resources. Most questions were yes/no or multiple choice, with several text boxes included to allow for comments from participants to provide additional information or clarification. Responses were analyzed by the proportion of affirmative responses, with common themes drawn from the subjective text responses. A copy of the questionnaire is included (Supplemental Appendix I) and Table I summarizes the key topics of the questions. Institutional review and university ethics approval were obtained prior to the initiation of this research project.

Table I. Summary of Topics in Questionnaire

Does the institution have a dedicated pediatric oncology follow-up program?

If yes, questions pertaining to the following were asked:

Duration of follow-up

Health professionals involved within institution

Access to resources

Location of clinic

Health professionals involved outside institution

Database maintenance

Funding

Presence of traveling outreach clinics

Does the institution have a formal program for adult survivors of childhood cancer?

If yes, questions pertaining to the following were asked:

Patient inclusion criteria for follow-up

Health professionals involved within institution

Access to resources

Database maintenance

Funding

Presence of traveling outreach clinics

RESULTS

Fifteen of 16 centers participated in the survey. Tables II and III summarize many of the findings. The total number of respondents varies with some questions as not all questionnaires were fully completed. Of the 15 participating centers, 13/15 (87%) had a formal long-term follow-up program, defined as a dedicated program for patients greater than 2 years beyond active treatment. Both of the two centers that did not have a formal program continued to follow survivors for late effects in their regular oncology clinics. One of these two centers has a formal late effects program in development. The directors of these clinics were pediatric oncologists in 12/14, or 86% of centers, with one blank response. Ten of 13 (77%) respondents indicated their center transferred patients to a separate long-term follow-up clinic (if one existed) by 5 years post-active therapy. Two respondents left the question blank.

Table II. Summary of Pediatric Late Effects Programs

Number of pediatric oncology centers with a formal long-term follow-up program

13/15 (87%)

Director of pediatric oncology long-term follow-up program

Pediatric Oncologist 12/14 (86%)

Number of pediatric oncology programs which follow survivors until 16th birthday

15/15 (100%)

Number of pediatric oncology programs which follow survivors until 25th birthday

8/15 (53%)

Health care professionals present in pediatric long-term follow-up clinics:

Pediatric oncologist 13/14 (93%)

Nurse 10/14 (72%)

Nurse Specialist 5/14 (36%)

Nurse Practitioner 2/14 (14%)

Psychologist 8/14 (57%)

Social Worker 6/14 (43%)

Timely access to services (affirmative responses)

13/15 (87%)

Other health care centers involved in pediatric late effects follow-up

General practitioners' offices 2/12 (17%)

General pediatric practices 3/12 (25%)

Outside follow-up clinics affiliated with long-term follow-up program (satellite)

5/12 (42%)

Traveling or outreach clinic present

2/15 (13%)

Table III. Summary of Programs for Adult Survivors of Pediatric Cancer

Number of Centers with Formal Programs for Adult Survivors of Childhood Cancer

8/15 (53%)

Number of programs in which 50% or more of survivors were included in program

4/10 (40%)

Director(s) of program

Pediatric oncologist 6/9 (67%)

Medical oncologist 2/9 (22%)

Radiation oncologist 3/9 (33%)

Nurse or nurse specialist 6/9 (67%)

Nurse practitioner 1/9 (11%)

Health professionals present in such clinics:

Pediatric oncologist 7/10 (70%)

Medical oncologist 2/10 (20%)

Radiation oncologist 5/10 (50%)

Nurse/nurse specialist 8/10 (80%)

Nurse practitioner 3/10 (30%)

Psychologist 4/10 (40%)

Social worker 5/10 (50%)

Timely access to services (affirmative responses)

5/10 (50%)

Traveling or outreach clinic

None

Almost all centers reported that referrals could be made to subspecialists such as pediatric endocrinologists, cardiologists, gynecologists, and respirologists if required. Most centers (12/14) followed patients yearly, with the remainder following patients every 2 years.

Nine of 15 centers (60%) maintained an information database for research purposes. Summaries of care were provided for survivors by 10/15 (67%) of centers. Funding was provided primarily from hospital and provincial health plan budgets, with support from the Pediatric Oncology Group of Ontario (POGO) identified by two centers. Donations were identified as funding sources in several programs.

When asked if their center has a formal program for adult survivors of childhood cancer, 8/15 (53%) of participants gave an affirmative response. Two of fifteen centers had such a program in development. Interestingly, only 4 of these 10 programs felt that 50% or greater of their adult survivors were followed in their formal program. In fact, several free text responses described concerns about attrition upon transfer of their patients to adult follow-up programs, and that no resources or mechanisms are in place to identify and contact survivors who have been lost to follow-up.

The most important determinants as to whether a childhood cancer survivor is followed in a formal adult late effects program were: existing late effects at the time of transfer, potential adult onset of late effects, and disease status (short time from completion of therapy). All programs had subspecialists available for referral. However, only 5/10 respondents felt investigations for adult survivors of childhood cancer could be obtained in an adequate and timely manner. Most adult follow-up programs for survivors of childhood cancer also had databases.

DISCUSSION

The field of pediatric oncology has seen tremendous improvements in survival of children with cancer over the last 40 years 1, 2. With increasing cure rates, care of survivors of childhood cancer is becoming a growing responsibility, and there remains much to be learned about the complications of the therapies used to achieve cures 7–10. With an increasing number of survivors, greater demands will be placed on existing resources to provide adequate follow-up care for this growing population 3, 5. Few studies have addressed the follow-up programs available for survivors of childhood cancer 1, 2, 5. Our hope was to demonstrate what services are in place in Canada, draw more attention to the growing practice of late-effects care and to emphasize the need for additional resources and research in this field.

While many pediatric centers continue to follow survivors for late effects, fewer dedicated programs for adult survivors of childhood cancer exist in Canada. Concerns about attrition of patients upon transfer of care seemed to be a universal concern among respondents. The loss of survivors to follow-up means that such patients will not be monitored for late effects, nor might they benefit from new research data. With continually emerging information in late effects research, it would be tragic for survivors to miss out on resulting opportunities for disease prevention and health promotion.

There were surprisingly few general practitioners, general pediatricians, general internists, and nurse practitioners involved in these programs nationwide, in contrast to some other models used internationally 2, 4, 5. We feel this is an area that requires attention, as many survivors have multiple medical issues, sometimes outside the scope of expertise of a pediatric oncologist. As previously mentioned, many pediatric oncologists continue to follow survivors well into adulthood, often due to the absence of a formal adult follow-up program. In addition, several respondents in our study mentioned reluctance from many practitioners to accept the transfer of care of these complicated patients, either due to discomfort or due to disinterest. It seems from our study that more complicated patients receive closer follow-up, however, this leaves clinically well survivors with known or potential risks related to their therapy without adequate surveillance. Perhaps greater involvement of primary care practitioners would allow for more comprehensive follow-up of adult survivors of childhood cancer, and such programs might improve the services available for such patients. Future research and program development for these adult survivors is essential, so as to decrease attrition rates, improve health surveillance and to capture more data about late effects in survivors as they age. Research and registry databases are essential in this regard.

Limitations of this study include many of the challenges inherent in questionnaire based research. Participants gave responses based on their knowledge of what programs were available in their respective centers. Subjective responses were clearly dependent on the individual participant. Not all respondents answered all questions. All but one center responded, which reduces concerns regarding self-selection bias.

The field of late effects continues to grow, and clearly Canadian pediatric and adult oncology programs have established much needed services for this unique population. Some new programs are in development, but there is still a need for more resources to meet the care needs of this growing population. The inclusion of more primary care practitioners in the care of survivors is essential, with continuing education of all those who provide care for these patients being paramount. Establishing mechanisms such as a registries for re-contacting patients lost to follow-up for various reasons (e.g., change of address) would perhaps address the major issue of attrition. The continued maintenance of research databases to allow for more clinical research in the field of late-effects should also be a priority. Formal long-term follow-up programs provide many benefits for survivors, including the maintenance of good health, surveillance for late effects of therapy and appropriate early intervention when complications arise.

Acknowledgements

Many thanks to the following institutions for their participation: British Columbia's Children's Hospital, Alberta Children's Hospital, Stollery Children's Hospital, CancerCare Manitoba, Children's Hospital of Western Ontario, McMaster Children's Hospital, Hospital for Sick Children, Kingston General Hospital, Children's Hospital of Eastern Ontario, Hôpital Sainte-Justine, McGill University Health Centre, Centre Hospitalier Universitaire de Sherbrooke, Centre Hospitalier Universitaire de Québec, IWK Health Centre, and the Janeway Children's Health and Rehabilitation Centre.