The unmet needs of childhood cancer survivors in long‐term follow‐up care: A qualitative study

Abstract Objective Evidence suggests benefits of long‐term follow‐up care attendance for childhood cancer survivors, but studies show poor inclusion of survivorship issues and needs. While information needs of childhood cancer survivors have been addressed previously, few studies specifically investigated the supportive care needs of survivors beyond the domain of information and communication. Therefore, this qualitative study aimed to assess the unmet needs of childhood cancer survivors with regards to their long‐term survivorship. Methods Childhood Cancer Switzerland invited survivors of childhood cancer to participate in our study. We used semistructured interviews to assess survivors' experiences regarding the impact of their disease and the (un)met needs during their survivorship. Data analysis followed the principles of qualitative content analysis. Results Interviews were conducted with 28 childhood cancer survivors (mean age 31 years, age at diagnosis 9 years, time since end of treatment 19 years). Key themes in relation to unmet needs of survivors were (1) lacking psychosocial support, (2) lacking collaboration and decentralization of care, (3) starting from zero, and (4) need for centralized, specialized, and individualized services. Conclusions Our findings revealed a demand for integrating psychosocial support in long‐term follow‐up care and a strong need for personalized, centralized, and interdisciplinary long‐term follow‐up care. Current established interdisciplinary clinics should be further extended to provide centralized, personalized, and evidence‐based long‐term follow‐up care including adequate psychosocial support for all childhood cancer survivors.


| BACKGROUND
Long-term follow-up care of childhood cancer survivors has received increasing attention since treatment improvements have led to a growing number of survivors over the past decades. 1 Currently, more than 80% of children diagnosed with cancer survive and ten-year survival has significantly improved (>87%). 2 However, with successful treatment, a life-long risk of adverse health effects for childhood cancer survivors arises 3 including medical and psychosocial issues. [4][5][6] To improve quality of life and diagnose treatable late effects early, providing high quality long-term follow-up care is of imminent importance. In fact, the goal of a timely long-term follow-up care is to prevent, detect, or decrease severity of late treatment effects through preventative therapeutic measures, health promotion, and psychosocial support. 7 Recently, the need to implement effective and efficient long-term follow-up models of care has become recognized in several countries. 8 In Switzerland, follow-up care has long been available in the first 5-10 years after end of treatment and most centers provide transition from pediatric to adult care (usually at 18-20 years). That is to say general practitioners or clinical oncologist provide adult childhood cancer survivors care, albeit mostly without detailed evidence-based information and instruction on long-term follow-up. Once transitioned into adult care, long-term follow-up is poorly standardized. [9][10][11] Currently, only four centers provide interdisciplinary long-term follow-up care with pediatric oncology for adult childhood cancer survivors. One of these centers focuses on childhood cancer survivors previously lost to long-term follow-up after transitioning into adult care. 12 More recently, some clinics have started to provide detailed survivorship care plans including treatment summaries as recommended by guidelines. 9,12 However, childhood cancer survivors and health care professionals (HCPs) in Switzerland and other countries continue to describe lacking long-term follow-up programs and report heterogeneity in aftercare among centers. 8,13,14 Evidence has shown poor inclusion of survivorship issues and needs. [15][16][17][18] It is important to address unmet needs as studies have shown that survivors with information needs experience more psychological distress and lower quality of life. [19][20][21] Especially, unmet information needs are known to negatively impact long-term follow-up attendance or seeking of medical care. 10,[19][20][21] While there is some evidence on childhood cancer survivors' information needs, 19,22 few studies specifically investigated the supportive care needs beyond the domain of information and communication. 23,24 However, childhood cancer survivors may experience other types of unmet needs. Recently, we have shown that alongside largely unmet informational needs, a subset of Swiss childhood cancer survivors further report unmet psychosocial needs. 25 In this study, we build on these quantitative results with an in-depth understanding of the unmet needs of childhood cancer survivors.
With the increasing number of childhood cancer survivors, optimizing long-term follow-up care is essential to provide efficient and effective, high-quality care for all survivors. 26,27 In turn, addressing childhood cancer survivors' needs might help increase their long-term follow-up attendance. 10,24,27 Thus, we aimed to assess the supportive care needs of childhood cancer survivors with regards to their long-term survivorship.

| METHODS
This paper builds on the results of a cross-sectional survey on the impact of cancer and childhood cancer survivors' unmet needs during survivorship. For the current study, we used a qualitative research design 28 and conducted semistructured interviews with a subset of participants from the survey. The Ethics Committee Northwest and Central Switzerland (Study-ID: EKNZ 2017-01758) approved the study.

| Participants and procedure
Childhood cancer survivors had been identified through Childhood Cancer Switzerland, the umbrella organization of childhood cancer associations in pediatric oncology in Switzerland. 25 Participants were eligible if they were aged ≥ 18 years at time of study, diagnosed with cancer ≤ 18 years of age, ended treatment ≥ 2 years before study, were Swiss residents, and spoke German or English.
Childhood Cancer Switzerland sent an e-mail invitation to all its registered survivors (n ¼ 132) to participate in a cross-sectional survey. We sent a reminder to nonresponders after 2 months. These survey participants were invited to take part in an interview in order to obtain a more nuanced understanding of their experiences, preferences, and needs regarding their long-term survivorship.

| Data collection
Survey participants who were interested in the qualitative study shared their contact information with the study team. Interviews were scheduled at a location selected by childhood cancer survivors (i.e. participants' home, workplace, or a meeting room at a University). The first author conducted all interviews between November 2017 and February 2019. The interviews were conducted until theoretical saturation was achieved. 28 Before the interviews, participants received written and oral information about the study. They

| Data analysis
Data analysis followed the principles of qualitative content analysis, which takes into account previously defined research questions outlined by the literature and allows categories to emerge out of the data. Two members of the research team (MJH and EH) reviewed the transcripts to identify emerging themes and together developed an initial coding frame utilizing a deductive-inductive procedure. 29 This approach integrates elements from grounded theory such as theoretical memos and iteration to generate descriptive results and conceptual models of the topics under study. 29 Hence, this approach is focused on the importance of context in determining meaning, that is data driven and iterative.
An initial coding scheme was developed based on our research question, interview guide, and reviewed literature. First, preliminary codes were generated through systematic coding of the data by MJH and EH. Second, identified codes were reviewed and refined.
Consensus for coding was reached. All transcripts were recoded at the end of the process using the finalized coding structure. 29 Qualitative data organization and aggregation were facilitated by Atlas.ti 8.3 (data available upon written request).
The most frequently reported diagnosis was leukemia (10/28), and mean time since end of treatment was 19 years ( Table 1). The key themes that emerged in relation to the unmet needs of childhood cancer survivors were (1) lacking psychosocial support, (2) lacking collaboration and decentralization of care, (3) starting from zero, and (4) need for centralized, specialized, and individualized services.

| Lacking psychosocial support
Regardless of time since end of treatment most childhood cancer survivors described how provision of psychological or psychosocial support was lacking. This means that psychological and social support was lacking in follow-up as well as in long-term follow-up. Childhood cancer survivors mentioned that it was difficult to get support from many services and "there is not really anything." Either no point of contact exists or the service is overwhelmed with requests. One childhood cancer survivor described how HCPs would merely ask whether he was "doing ok" with a focus on his physical wellbeing while offering "zero psychological support." In addition, this burden is further exacerbated when there is limited information or faulty communication channels for finding formal support. Survivors reported feelings of "being lost." One survivor described how during her appointment, even her HCP could not provide further information on where to find psychological support.

| Lacking collaboration and decentralization of care
Furthermore, many childhood cancer survivors described a lack of interdisciplinary collaboration amongst HCPs. They reported having several different HCPs from different disciplines that they visit monthly or yearly for their long-term follow-up care. However, sometimes communication and collaboration between these disciplines was lacking, which caused distress. As one survivor put it; "it is every department for itself," in which quality curtails when everyone is working only within the limitations of their own medical field.
Another survivor explained how the lack of collaboration lead to "disorganized long-term follow-up care" such that in the end she often did not know to whom she could go with questions regarding her care.
Not only did childhood cancer survivors consider long-term follow-up as lacking a well-established structure for collaboration, survivors also mentioned the (dis)stress from decentralized care.
Survivors reported having many different appointments on different days in different locations with different HCPs. All survivors considered long-term follow-up appointments to be a loss of their (economic) productivity and a great organizational effort. One childhood cancer survivor reported having had five to seven appointments in one week. This meant she had to travel to two different hospitals to visit her two specialists. In turn, she missed school, and had to additionally justify her absence.

| Need for centralized, specialized, and individualized services
In light of the above, a majority of childhood cancer survivors called for a central resource or "point of contact." This would unite several services or would provide coherent information on where to find such services (Table 2). Furthermore, they wanted centralized longterm follow-up care provided by specialized HCPs with knowledge on survivorship. As such, "a centralized interdisciplinary" service was considered "the first priority" to improving long-term follow-up for survivors.

| DISCUSSION
In this qualitative study, we found that even long after the end of treatment childhood cancer survivors have many unmet needs. Survivors reported lacking psychosocial resources and support in longterm follow-up care and expressed a strong need for centralized T A B L E 2 Points of contact for long-term follow-up care

Types Quotes
Centralized It would have really helped to have someone who could figure it out, for whom perhaps it would also not take so much time and effort. Such a person could have really taken care of the situation. But I do not think such a person exists. I Think that is really something to strive for. Really someone expert on childhood cancer specific concerns, so someone can really support you.-Male survivor, ≥ 25 years since end of treatment, late effects (interview #3M) Psychological So I think, when you are really acutely ill and I mean, if you already say, I need help, but that you then have to wait 3,4 weeks for an appointment just because you are not suicidal, I think that is quite strange […] Yes, I think it would actually be good if there was like a point of contact, like an emergency phone number, where you can call and that they would just quickly and at any time have an appointment where you can really go, because really, I think it's insane, that you have to wait so long and I mean in this state I couldn't go to work but I knew I wanted to go back to work soon because it would be good for me if I could go to work and then just sit at home for a month and just simply be, well that startled me pretty much I have to say. -489 long-term follow-up care centers. In recent years, four interdisciplinary long-term follow-up clinics have opened in Switzerland with one centralized clinic for long-term survivors who were lost to longterm follow-up. 12 To meet the needs of this growing population, longterm follow-up care for all Swiss childhood cancer survivors needs to be optimized. 8,12,14 Despite standards of care recommending provision of psychosocial support for childhood cancer survivors also in long-term

| Clinical implications
To meet the needs of survivors it is essential to provide easy access to long-term follow-up care for all childhood cancer survivors. Two recommendations can be made on the basis of our findings. First, more centralized interdisciplinary clinics should be developed. Second, psychosocial services should be implemented as part of longterm follow-up care, which is in accordance to international recommendations. 1

| CONCLUSIONS
Our study showed that psychosocial support in long-term follow-up care played a crucial role in childhood cancer survivors' unmet needs and should be included in future survivorship care. 12 In addition, survivors expressed a strong need for centralized interdisciplinary long-term follow-up clinics. Lifelong specialized survivorship care and 490ongoing late effects education for survivors will help to improve health and mental wellbeing. 39 We, therefore, recommend to include psychosocial services in long-term follow-up together with the development of personalized and evidence-based long-term followup for survivors, and especially for very long-term survivors after childhood cancer.