Perception Gap in Health-Related Quality of Life Between Young Adult Survivors of Childhood Cancer and Their Family

Abstract

Young adult survivors of childhood cancer may have a perception gap with their families. Patients aged 18–39 years after treatment of cancer and their families (28 pairs) completed a survey that contained questions on health-related quality of life using the 36-item short form survey. There was a significant difference in the role-social component score (mean difference −2.23; p = 0.04) with family reporting higher scores than patients. Families may overestimate the social function of cancer survivors, emphasizing the importance of the long-term follow-up by taking into account the risk of a gap (IRB approval No.: R2257-1).

Introduction

Cancer is no longer a deadly disease but a chronic one for childhood cancer survivors in most cases. In Japan, the conditional 5-year survival of 5-year survivors has been estimated at >94% (male: 94.9%, female: 96.8%) for all children (aged 0–14 years) with cancer.¹ As survivors of childhood cancer become adolescents and young adults, they may face unique challenges due to their condition. A wide range of late physical and psychological effects are reported in adolescent and young adult survivors (AYAs), emerging long after the end of childhood cancer treatment.^2–4

Studies have identified the importance of age-appropriate support for AYAs.^5–7 AYAs undergo complex physical and psychosocial development with substantial lifestyle changes. Adolescents often leave home for school, begin working, and become more independent from their parents. Young adults tend to prioritize completing their education, securing stable employment, getting married, and having children. A systematic review highlighted the need for age-appropriate care for AYAs, including fertility information, peer contact, emotional support, and support for psychosocial well-being.⁵ Studies indicate that that needs change as AYAs fight their way through diagnosis and active treatment and into survivorship.^6,7 Another study reported the importance of age-appropriate care for AYAs, for whom care is entirely divided into childhood and adulthood.⁸ A systematic review highlighted the importance and potential for long-term follow-up care.⁹ After treatment, long-term support for their needs is warranted.

For those childhood cancer survivors who undergo various changes in their lifestyle, family support is essential. A previous study reported that the role of family and friends seems to be primary for both patients in-treatment and off-treatment survivors.¹⁰ Cancer patients have different needs according to their age, diagnosis age, and time since diagnosis.¹⁰ Moreover, the family is the principal decision maker regarding the patients' lifestyle, even after childhood. Thus, understanding the family's perceptions of patients' health-related quality of life (HRQOL) and comparing them with those of the patient will have important implications for treatment decisions.¹¹

Studies have reported a perception gap in HRQOL between patients and their families.^11–13 There is a tendency for the family of patients with a serious illness to underestimate the HRQOL of the patients on various domains, including mental health, general HRQOL, role limitations, and physical functioning.^14–16 This tendency to report lower levels of HRQOL exists for both children and adolescents.¹⁵ However, the perception gap between long-term childhood cancer survivors and their families remains unclear. This gap can cause families to overlook patients' late effects and limitations to activity; therefore, further research on it is necessary. Accordingly, this study examined the patient–family perception gap in reports of HRQOL in young adult survivors of childhood cancer.

Methods

Participants and procedures

This study examined the gap between cancer patients and their families regarding patients' HRQOL. The study consisted of a cross-sectional questionnaire survey of young adult survivors of childhood cancer and their families. Participants of cancer survivors were included if they were between the ages of 18 and 39 years after treatment. Patients who lived alone were excluded. Family members in our study included parents and spouses who lived with patients in the same house. Data collection was conducted between July 2020 and October 2020. We recruited participants at Kyoto University Hospital Pediatric Department and handed or sent the questionnaire to 63 patients and families who agreed to participate. The patient's version of the questionnaire including 36-item short form survey (SF-36) asked questions about the patients' HRQOL. The family's version of the questionnaire asks about the patients' HRQOL from the family's perspective. These questionnaires took ∼10 minutes to complete. We obtained the demographic and clinical data of patients from electronic health records. Ethical approval was granted by the ethics committee of Kyoto University Graduate School and Faculty of Medicine.

Measures

HRQOL was measured using the Medical Outcomes Study Short Form-36 (SF-36) Health Survey. The SF-36 is a commonly used and well-validated instrument that assesses generic HRQOL¹⁷ and measures HRQOL in eight domains: physical functioning, role physical, bodily pain, general health, vitality, social functioning, role emotional, and mental health. Scoring through t-scores using the Japanese general population means and standard deviations and weighting produces three component scores: physical, mental, and role social.¹⁸ Role physical, role emotional, and social functioning had strong loadings on the role social component score.¹⁸ These summary scores ranged from 0 to 100, with higher scores representing better quality of life. Family and patient questions for the SF-36 were identical, except for patients being asked about “your” health and proxies being asked about “patient's” health.¹⁹

Data analysis

Descriptive analyses

Means, standard deviations, and proportions were used for descriptive analyses.

SF-36 mean difference

A Wilcoxon signed-rank test was performed to examine gaps in HRQOL between patients and their families. Spearman's signed-rank test was performed to examine the correlation between patient-reported and family-reported scores. All statistical tests were two sided and considered significant if p < 0.05. All analyses were conducted using JMP Pro 15 (SAS Institute, Inc.).

Results

The demographic and clinical characteristics of 56 participants (28 pairs) who completed the surveys are summarized in Table 1. We found 68% of patients experienced acute lymphocytic leukemia and 71% of patients received chemotherapy.

Table 1.

Sociodemographic and Clinical Characteristics

Characteristics	Patients	Family
Sociodemographic characteristics
Age (years), n
18–23	16
24–28	4
29–33	5
34–39	3
30–39		3
40–49		3
50–59		4
60–69		1
Uncertain		17
Gender, n
Female	14	21
Male	14	7
Employment/school status, n
Not occupied	3	8
Occupied	24	20
Uncertain	1
Relationship with patients, n
Mother		20
Father		3
husband		4
Wife		1
Clinical characteristics
Age (years) at diagnosis, n
0–9	11
10–19	15
uncertain	2
Time since treatment, n
5< years	2
5–10 years	6
10–15 years	9
15–20 years	7
20< years	4
Type of cancer, n
Acute lymphocytic leukemia	19
Acute myeloid leukemia	4
Non-Hodgkin lymphoma	1
Brain tumor	1
Ovarian cancer	1
Hodgkin lymphoma	1
Uncertain	1
Type of treatment, n
Chemotherapy	19
Transplant	4
Chemotherapy and transplant	3
Uncertain	2

Table 2 summarizes the SF-36 results for both patients and the patient-by-family. Although there was no significant difference in physical and mental domains, patient-reported HRQOL was significantly lower than family-reported HRQOL in the social component score (mean difference −2.23; p = 0.04), especially vitality (mean difference: 4.50; p = 0.04), role emotional (mean difference −2.92; p = 0.03), and social functioning scores (mean difference −2.68; p = 0.001). In the correlation analysis, the mental component score (ρ = 0.52; p = 0.01), physical functioning (ρ = 0.71; p < 0.0001), general health (ρ = 0.38; p = 0.04), vitality (ρ = 0.63; p = 0.0004), and mental health (ρ = 0.41; p = 0.03) exhibited a significant correlation between patients and their families.

Table 2.

Agreement of Patient and Patient-by-Family Ratings for 36-Item Short Form Survey

SF-36	Mean patient	Mean family	Mean difference	p ^a	ρ^b	p ^b
Summary score
Physical component score	53.2	50.9	2.30	0.91	0.22	0.26
Mental component score	54.6	55.6	−1.00	0.27	0.52	0.01^*
Role social component score	50.4	53.2	−2.80	0.04^*	−0.06	0.77
Subscale
Physical functioning	92.1	93.2	−1.10	0.35	0.71	<0.01^*
Role physical	92.0	92.4	−0.40	0.53	0.33	0.09
Bodily pain	87.9	79.1	8.80	0.90	0.14	0.47
General health	66.6	69.6	−3.00	0.12	0.38	0.04^*
Vitality	65.6	70.1	−4.50	0.04^*	0.63	<0.01^*
Social functioning	92.0	94.6	−2.60	0.03^*	0.08	0.67
Role emotional	51.1	54.0	−2.90	<0.01^*	0.37	0.06
Mental health	73.8	77.0	−3.20	0.23	0.41	0.03^*

Wilcoxon signed-rank test.

Spearman's correlation coefficient.

p < 0.05.

SF-36, 36-item short form survey.

Discussion

The main finding of this study was that the perception gap in HRQOL between young adult childhood cancer survivors and their families existed in the social domain, where families overestimated the patients' HRQOL. Although we found that there was overall high agreement between patients and family reported in the physical and mental subscales of the SF-36, statistically significant differences were observed in the role social component, social functioning, and role emotional scores.

Past research suggests that families tend to underestimate patients' HRQOL when compared with the patients' reports.^11–13 Concerning childhood cancer patients, studies have reported that parents underestimate children's HRQOL.^11–13 Studies on adult cancer patients indicate that families underestimated patients' HRQOL and symptoms such as pain, depression, anxiety, drowsiness, and loss of well being.^19,20 The family's underestimation of HRQOL is also reported in other chronic diseases, such as pediatric asthma.^13,21

Although these studies demonstrate the tendency of families to underestimate patients' HRQOL, in this study, families overestimated the patient's social aspect of HRQOL. This is partly because the time since the treatment of the participants in the previous studies^11,12,19 was 1–5 years, whereas most participants in our study were treated for >10 years. The overestimation of patients' HRQOL has been found in a previous study of healthy samples.¹¹ Families could gradually take survivors as noncancer patients and overestimate survivors' HRQOL. As survivors got older, they expressed more treatment anxiety while families believed the survivors' treatment anxiety reduced as they aged.²² These results suggest that longer follow-up periods may cause families to overlook patients' late effects.

A significant difference was found in the role social domain. From a perspective of families, a previous cohort study showed that chronic medical conditions after cancer therapy increase the risk for unemployment,²³ and another study reported that families of childhood cancer survivors feel anxious about survivors' employment and social relationships.²⁴ In our study, 86% of patients had a job or went to school. It appears that when childhood cancer survivors are gradually getting used to their normal lives, their families may get relieved and overestimate their condition.

Considering survivors' circumstances, although they adapt to their physical and social limitations through childhood into adolescence and young adulthood, they are often confronted with late effects. Late effects may persist not only during treatment or after several years but also decades later.⁴ Physical symptoms could have a harmful effect on survivors' social skills and activities, such as educational and vocational disadvantages and infertility.^22,25 This perception gap may lead to the families' overestimation of survivors' social HRQOL.

A systematic review reported that there was a greater agreement for visible functioning (e.g., physical HRQOL) and less for invisible functioning (e.g., emotional or social HRQOL).²⁶ Although most studies in the review reported the tendency of family underestimation, the family overestimation in social HRQOL, which was found in our study, could also be attributed to the visibility. In particular, it has been reported that the percentage of families who perceived fertility as a late effect was 20% lower than that of survivors.²² This overlook may cause families to be unable to provide necessary support.

These results from the comparison of patient–family reports may have important implications for the understanding of long-term childhood cancer survivors. Most studies only focus on individuals for the evaluation of the survivors. However, the family is one of the most essential sources of support even after treatment. A previous study on pediatric asthma patients²¹ reported that the extent and direction of disagreement were better explained by family-related factors than by sociodemographic and clinical variables such as family relationships. Future studies should focus on not only the patients themselves but also the whole unit of patient and family.

Several limitations should be acknowledged in this study. The first is selection bias. We did not have any restriction for survivors on time from diagnosis because we aimed to examine widely the perception gap between long-term survivors of childhood cancer and their family. We restricted only the current age up to 39 years old and only included family members who lived with participants in the same house and did not have any more criteria in terms of feasibility and diversity. We expected to correct questionnaires more successfully if participants lived in the same house. Although it was ideal to correct questionnaires from families who lived apart from survivors, we made it a priority to include widely those who were close to the survivors and lived in the same house regardless of blood or marital relationship. In our samples, there were 20 mothers, 3 fathers, 4 husbands, and 1 wife. The second is the small sample size. Although our samples had variation in age, time from diagnosis, and the relationship between survivors and families, we were unable to examine the statistical difference by each characteristic (i.e., the t-scores for SF-36 are not age adjusted) due to the small sample size. The replication in larger samples is warranted to investigate the perception gap in more detail. Third, the evaluation consisted of general HRQOL and did not include disease-specific questionnaires. To investigate the influences of disease, ages, time points, and family members, further large-scale studies with various background participants are needed.

This is the first study to our knowledge to investigate the patient–family perception gap in reports of HRQOL in young adult childhood cancer survivors, demonstrating that families can overestimate patients' HRQOL when a long time period has passed since treatment. The overestimation by family members was reported only within healthy samples, and thus our results provide significant information on the life-long consequence of survivors of childhood cancer. Our findings emphasize not only the importance of the family's lifelong support to the needs of cancer survivors but also the possibility that patients and families may have some perception gap in the patient's condition. Therefore, health care providers should provide long-term follow-up by taking into account the risk of a gap.

Footnotes

Acknowledgments

The authors thank R. Goi for data collection with this study and gratefully appreciate the participation of the survivors of childhood cancer.

Author Disclosure Statement

No competing financial interests exist.

Funding Information

No funding was received.

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