Received: 31 January 2021 Revised: 24 August 2021 Accepted: 11 September 2021

DOI: 10.1002/pbc.29398 Pediatric

Blood &
Cancer The American Society of
Pediatric Hematology/Oncology
SURVIVORSHIP: RESEARCH ARTICLE

Long-term health-related quality of life in young childhood

cancer survivors and their parents

Joanna E. Fardell1,2,# Claire E. Wakefield1,2 Richard De Abreu Lourenco3

Christina Signorelli1,2 Maria McCarthy4,5 Jordana McLoone1,2
Michael Osborn6 Melissa Gabriel7 Antoinette Anazodo1,2,8 Frank Alvaro9
Liane Lockwood10 Thomas Walwyn11 Jane Skeen12 Ramon Tillemans2,3
Richard J. Cohn1,2 ANZCHOG Survivorship Group1,#
1
Kids Cancer Centre, Sydney Children’s Hospital, Randwick, New South Wales, Australia
2
School of Women’s and Children’s Health, UNSW Sydney, Kensington, New South Wales, Australia
3
Centre for Health Economics Research and Evaluation, University of Technology Sydney, Haymarket, New South Wales, Australia
4
Children’s Cancer Centre, Royal Children’s Hospital, Melbourne, Victoria, Australia
5
Clinical Sciences, Murdoch Children’s Research Institute (MCRI), Melbourne, Victoria, Australia
6
Paediatric Haematology/Oncology, Women’s and Children’s Hospital Adelaide, North Adelaide, South Australia, Australia
7
Cancer Centre for Children, The Children’s Hospital at Westmead, Sydney, New South Wales, Australia
8
Nelune Comprehensive Cancer Centre, Prince of Wales Hospital, Sydney, New South Wales, Australia
9
John Hunter Children’s Hospital, Faculty of Health and Medicine, University of Newcastle, Newcastle, New South Wales, Australia
10
Oncology Service Group, Children’s Health Queensland, Brisbane, Queensland, Australia
11
Perth Children’s Hospital, School of Paediatrics & Child Health, University of Western Australia, Perth, Western Australia, Australia
12
Starship Blood and Cancer Centre, Starship Children’s Hospital, Auckland, New Zealand

Correspondence
Joanna Fardell, Kids Cancer Centre, Level 1 Abstract
South, Sydney Children’s Hospital, High St,
Randwick, NSW 2031, Sydney, Australia.
Purpose: Few studies have investigated the health-related quality of life (HRQoL) of
Email: [email protected] young childhood cancer survivors and their parents. This study describes parent and
child cancer survivor HRQoL compared to population norms and identifies factors
Funding information
The Kids’ Cancer Project; Career Devel- influencing child and parent HRQoL.
opment Fellowship (NHMRC of Australia),
Methods: We recruited parents of survivors who were currently <16 years, and
Grant/Award Number: APP1143767; Kids with
Cancer Foundation; Kids Cancer Alliance; The >5 years postdiagnosis. Parents reported on their child’s HRQoL (Kidscreen-10), and
Kids’ Cancer Project; Cancer Council NSW Pro-
their own HRQoL (EQ-5D-5L). Parents rated their resilience and fear of cancer recur-
gram, Grant Number: PG16-02; Estate of the
Late Harry McPaul; Victorian Governments’ rence and listed their child’s cancer-related late effects.
Operational Infrastructure Support Program;
Results: One hundred eighty-two parents of survivors (mean age = 12.4 years old and
Cancer Australia’s Support for Cancer Clinical
Trials. 9.7 years postdiagnosis) participated. Parent-reported child HRQoL was significantly
#
The members of the ANZCHOG Survivorship
lower than population norms (48.4 vs. 50.7, p < .009). Parents most commonly reported
Study Group in alphabetical order: Frank that their child experienced sadness and loneliness (18.1%). Experiencing more late
Alvaro, Richard Cohn, Rob Corbett, Peter
effects and receiving treatments other than surgery were associated with worse child

Abbreviation: HRQoL, health-related quality of life

Pediatr Blood Cancer. 2021;68:e29398. wileyonlinelibrary.com/journal/pbc © 2021 Wiley Periodicals LLC 1 of 10

https://doi.org/10.1002/pbc.29398
2 of 10 FARDELL ET AL .

Downie, Karen Egan, Sarah Ellis, Jon Emery,

Joanna Fardell, Tali Foreman, Melissa Gabriel, HRQoL. Parents’ average HRQoL was high (0.90) and no different to population norms.
Afaf Girgis, Kerrie Graham, Karen Johnston,
Janelle Jones, Liane Lockwood, Ann Maguire,
However 38.5% of parents reported HRQoL that was clinically meaningfully different
Maria McCarthy, Jordana McLoone, Francoise from perfect health, and parents experienced more problems with anxiety/depression
Mechinaud, Sinead Molloy, Lyndal Moore,
Michael Osborn, Christina Signorelli, Jane
(43.4%) than population norms (24.7%, p < .0001). Worse child HRQoL, lower parent
Skeen, Heather Tapp, Tracy Till, Jo Truscott, resilience, and higher fear of recurrence was associated with worse parent HRQoL.
Kate Turpin, Claire Wakefield, Jane Williamson,
Thomas Walwyn, and Kathy Yallop.
Conclusions: Parents report that young survivors experience small but significant
ongoing reductions in HRQoL. While overall mean levels of HRQoL were no differ-
ent to population norms, a subset of parents reported HRQoL that was clinically
meaningfully different from perfect health. Managing young survivors’ late effects and
improving parents’ resilience through survivorship may improve HRQoL in long-term
survivorship.

KEYWORDS
childhood cancer survivors, health-related quality of life (HRQoL), parents, psychological func-
tioning, resiliency, survivorship

1 INTRODUCTION socio-economic backgrounds.9–12 Particular domains of functioning

appear to be vulnerable in the years after treatment for paediatric can-
Improvements in treatment protocols mean that more children are sur- cer patients. Several studies have shown that psychological-emotional
viving cancer and living longer.1 However, life-saving cancer treatment functioning and social functioning are two particular domains of life
results in heightened risks of mortality and morbidity during survivor- that continue to be impacted long after treatment completion.9,13,14
ship. Large cohort studies indicate that survivors of childhood cancer Paediatric oncology has long recognised the interrelatedness of par-
are at increased risk for second cancers, cardiovascular disease, pul- ent and child adjustment to cancer, and accordingly family-centred psy-
monary disease, and psychological morbidity.1,2 Studies of the preva- chosocial care is considered fundamental.15 Parents’ well-being has
lence of chronic health conditions indicate that between 75% and 98% the capacity to impact their child’s well-being and family functioning
of survivors will experience at least one late effect of treatment as through cancer treatment and beyond.12,14,16 For example, children of
young adults, and that the risk of experiencing late effects and second parents who experience more distress and react more to stress, self-
cancers increases as survivors age.2,3 Understanding the true burden report a significant impact on their own HRQoL.10,12 Conversely, a
of surviving childhood cancer is paramount to better tailoring health child’s well-being can impact parents’ well-being. For example, when
and psychological care throughout survivorship.4 their child experiences more negative treatment side effects, greater
Quality of life is a subjective rating that an individual makes regard- treatment intensity and disease severity, parents report worse reduced
ing their overall functioning. Quality of life encompasses the domains psychological well-being and functioning.15
of physical, psychological, social and role functioning,5 and can also In the years after treatment completion, many families adjust to a
encompass functioning across other domains including spirituality and ‘new normal’; however, a subset continue to experience significant psy-
family functioning.6 Health-related quality of life (HRQoL) additionally chological distress, which impacts their HRQoL.16,17 Mothers, parents
considers functioning in the context of an individual’s health and illness with existing mental health concerns, and parents of children with dif-
symptoms.7 Assessment of HRQoL in cancer survivors allows the quan- ficulties adjusting to cancer diagnosis and treatment are at increased
tification of functioning during survivorship years. Identification of fac- risk of poor HRQoL.15,16 Parent appraisals of their own social and emo-
tors associated with worse HRQoL outcomes can inform practice and tional coping resources may be important determinants of HRQoL dur-
assist the development of services that are in line with survivors’ needs ing survivorship. For example, parents of long-term survivors of brain
as they age. tumours (10 years postdiagnosis) reported worse mental and physi-
Previous reviews suggest that while childhood cancer patients’ cal HRQoL when they perceived themselves as having fewer emotional
HRQoL is poor during treatment, survivors often report overall HRQoL resources and their child had poor peer relationships.18 The identifica-
at par with their peers.8,9 However, a subset of survivors appear to tion of risk factors is critical to preventing sustained poor HRQoL for
be at risk of reduced HRQoL. Survivors who were diagnosed with both parents and young survivors.
central nervous system (CNS) tumours and treated with more inten- Research has described the psychosocial outcomes of adult sur-
sive therapies (e.g., cranial radiation, transplant) report poor HRQoL, vivors of childhood cancer. Yet, few studies have specifically consid-
as do survivors from families with poor family functioning and low ered the HRQoL of young survivors who are still under the care of their
FARDELL ET AL . 3 of 10

parents. Given most childhood cancers are diagnosed in the first year of diagnosis, sex, whether they were attending a long-term follow-
5 years of life, the foundation for future well-being during survivorship up clinic for survivorship care, and how satisfied they were with this
is critically influenced by parents. Therefore, this study aimed to care (five response options ranging from ‘very unsatisfied’ to ‘very sat-
isfied’). Parents self-reported their current place of residence, level of
1. Describe the HRQoL of young survivors of childhood cancer and education and relationship to the child (e.g., mother, father). We coded
their parents, as compared to normative data, and place of residence into major city (metropolitan) or rural and regional
2. Identify factors associated with HRQoL among survivors of child- using the Accessibility Remoteness Index of Australia (ARIA) classifica-
hood cancer and their parents, which may indicate who is at risk of tion for Australian participants and the New Zealand Area Codebook
reduced HRQoL. for New Zealand participants.
Parent quality of life was assessed using the EQ-5D-5L, a validated
quality-of-life measure with five items assessing mobility, self-care,
2 METHODS ability to participate in usual activities, pain/discomfort, and anxi-
ety/depression; answered on a Likert scale with response options rang-
2.1 Participants ing from ‘no problems’ to ‘I am unable to’.7,19 A visual analogue scale2
regarding their overall health was also included asking participants
Eligible participants were parents of children (aged <16 years) who to rate their health today on a scale of 0–100, where 0 is the worst
were >5 years post any cancer diagnosis, had completed active treat- possible health they can imagine and 100 is the best possible health
ment, and were alive and in remission. One parent per child was eligi- they can imagine. The first five items are analysed together to deter-
ble to respond. We asked staff at the 11 paediatric hospitals in Aus- mine a quality-of-life index value, while the last indicates current health
tralia and New Zealand to identify a random sample of around 200 perceptions. Index values range from 0 to 1 (0 = death, 1 = perfect
potential participants who were <16 years of age when diagnosed, health). We calculated the EQ-5D-5L index using the UK population
and were now >5 years post any cancer diagnosis and had completed value set to enable comparison to an Australian normative popula-
active treatment through medical records. We then verified survivors’ tion, which also employed the UK value set in calculating HRQoL index
vital status with the Births, Deaths and Marriages Registry in Aus- scores.20 The Australian normative data are derived from 2908 adults
tralia and New Zealand (based on treating centre). After removing any aged over 15 years of age.20 We also calculated the proportion of par-
deceased individuals, we checked contact details with Australian and ents reporting any domain-specific difficulties according to developer
New Zealand residential directories. We expected a high number to instructions.21
be uncontactable given that they were discharged from their hospi- Parent-reported child quality of life was assessed using the Kidscreen-
tal prior to the study. We excluded parents with insufficient English to 10, a validated parent proxy measure of their child’s global HRQoL.22
complete the questionnaire and those deemed inappropriate to con- This 10-item scale addresses functioning in emotional well-being,
tact by their oncologist (e.g., due to severe psychosocial challenges that social functioning, activity levels, school performance, and quality of
would impair participation, e.g., psychosis). relationships with parents/caregivers and friends. Responses are on a
Likert scale and range from ‘not at all/never’ to ‘extremely/always’. We
summed scores across items, assigned Rasch person parameters and
2.2 Procedure then transformed these values into a T-score with a mean of 50 and a
standard deviation of 10 according to the Kidscreen Manual.23 Higher
We mailed potential participants a study pack that included an invita- scores indicate better HRQoL. We also calculated the proportion of
tion letter from the lead clinician at the hospital at which they were parents reporting item-specific problems for their child (0 = not at
treated, consent form, questionnaire (and link to online version of all/never a problem, 1 = any frequency level of problem). We used pub-
the questionnaire), and postage-paid envelope. We followed up nonre- lished norms derived from 16,237 parents who provided Kidscreen-10
spondents via telephone after 4 weeks, up to four times and re-sent the proxy responses.23
questionnaire up to two times. We prevented responses from duplicate Late effects were measured using a purpose-designed question. We
IP addresses, and all responses to the questionnaire were anonymous. asked parents to indicate (yes/no) on a list of late effects all the
The study was approved by the relevant ethics authority for each hos- health issues that they believed their child had experienced associ-
pital (Lead site reference 12/173 12/POWH/345) and was endorsed ated with their child’s cancer or treatment that had occurred since
by the Australian and New Zealand Children’s Haematology Oncology finishing treatment (see Supporting Material). We summed the num-
Group. Data available on request due to privacy/ethical restrictions. ber of late effects to get the total number survivors were currently
experiencing.
Fear of cancer recurrence and fear of late effects were assessed using
2.3 Measures two purpose-designed questions. We asked parents to indicate how
worried or anxious they were about their child experiencing a cancer
Clinical and demographic factors were reported by parents and included recurrence or late effects on a scale with five response options ranging
their child’s diagnosis, treatment type received, child’s current age, from ‘not at all’ to ‘a great deal’.
4 of 10 FARDELL ET AL .

Parent resilience was measured using the two-item Connor– TA B L E 1 Parent and child demographics
Davidson Resilience Scale (CD-RISC2).24 The two items assess
N (%)
whether the participant is ‘Able to adapt to change’, and ‘Tends to
Parent demographics (n = 182)
bounce back after hardship or illness’. Responses range from ‘not at all’
Fathersa 25 (14.5)
to ‘nearly all of the time’.
Education levelb
High school only 45 (24.7)

2.4 Data analysis Further education (including university 135 (74.2)

and other training)

We used IBM SPSS (IBM Corp, Version 22.0, Armonk, NY) for statis- Place of residencec
tical analyses. We used means, standard deviations and proportions Metropolitan 124 (68.1)
where appropriate to describe our sample. For Aim 1, we used two- Regional or remote 39 (21.4)
tailed Student’s t-tests to compare the HRQoL index and VAS on the Incomed
EQ-5D-5L to Australian normative data.20 We were unable to do
Less than $60,000 p.a. 37 (20.3)
agewise comparisons as we did not collect parent age, so we used the
More than $60,000 p.a. 126 (69.2)
overall mean Index score. We used chi-square goodness-of-fit tests to
e
Employment
compare the proportion of participating parents endorsing problems
Not currently employed 35 (19.2)
or not across the five domains of HRQoL as measured by the EQ-5D-
5L. We considered a reduction of 0.08 on the HRQoL and 7 on the VAS Currently employed (including full- and 144 (79.1)
part-time work)
to be indicative of minimally important difference (MID) in HRQoL.25
Marital status
For parent-reported child HRQoL, we compared the Kidscreen-10
T-score to published norms,23 using two-tailed Student’s t-test. We Not currently married/defacto 25 (13.7)

also considered the rate at which parents reported problems for their Currently married/defacto 157 (86.3)
child across the different items of the Kidscreen-10 using Cochrane’s Child demographics
Q test, with a Bonferroni correction for pairwise comparisons. Male 99 (54.4)
For Aim 2, to identify factors associated with HRQoL for parents of Age, current years (mean, SD) 12.4 (2.2)
childhood cancer survivors (as measured by the EQ-5D-5L index score)
Range 7–15
and parent-reported child HRQoL (as measured by the Kidscreen-
Time since diagnosis, years (mean, SD) 9.7 (2.1)
10 total score), we conducted a series of simple linear regressions
Range 5–15
for continuous and dichotomous variables, and one-way ANOVA for
categorical variables with more than two categories. We considered Diagnosis

child clinical variables, total number of late effects reported, parent ALL and AML 82 (45.1)
socio-demographic variables, parent fear of cancer recurrence and Brain cancer 9 (4.9)
late effects, and parent resilience. We coded child diagnosis accord- Lymphomas 13 (7.1)
ing to International Classification of Childhood Cancer (ICCC) into Neuroblastoma 16 (8.8)
leukaemia, lymphoma, brain cancer and other. We also included child
Sarcomas (bone and soft) 10 (5.5)
HRQoL when considering parent HRQoL and vice versa. We included
Wilms tumour 16 (8.8)
variables from the univariate analyses of parent and child HRQoL with
Other 36 (19.8)
p < .2 in the subsequent multivariable analyses. Results were consid-
Treatment
ered significant if p < .05.
Surgery 76 (41.8)
Chemotherapy 170 (93.4)
3 RESULTS Radiotherapy 46 (25.3)
Transplant 33 (18.1)
We approached 308 parents of childhood cancer survivors and
Abbreviations: ALL, acute lymphoblastic leukaemia; AML, acute myeloid
recruited 182 parents (59.1% response rate). Parent participants were leukaemia.
mostly mothers (80%) of childhood cancer survivors who were on aver- a
Missing 11.
b
age 12.4 years old (range 7–15 years) and 9.7 years since diagnosis Missing 2.
c
Missing 19.
(range 5–15 years; Table 1). Just over a half of survivors were male d
Missing 19.
(54.2%), many had survived leukaemia (46.4%) and the majority were e
Missing 3.
treated with chemotherapy (94.9%). There was no difference between
participating and nonparticipating parents’ residential location or child
characteristics of age and sex (p > .05).
FARDELL ET AL . 5 of 10

F I G U R E 1 Childhood cancer survivor health-related quality of life (HRQoL). In post hoc Bonferroni corrected pairwise comparisons, *parents
were more likely to report their child felt sad (18.1%) than having problems with having fun with friends (8.3%, p = .016), paying attention (7.7%,
p = .007), parents treated them fairly (6.6%, p = .003), got on well at school (6.6%, p = .003), was fit and well (6.6%, p = .001), being able to do the
things they want (2.2%, p < .001), had enough time for themselves (2.2%, p < .001). #Parents were more likely to report their child felt lonely
(18.1%) than having problems with having fun with friends (8.3%, p = .007), paying attention (7.7%, p = .003), parents treated them fairly (6.6%,
p = .001), got on well at school (6.6%, p = .001), was fit and well (6.6%, p = .001), being able to do the things they want (2.2%, p < .001), had enough
time for themselves (2.2%, p < .001)

On average, parents reported that survivors had experienced 3.2 rating for ‘felt fit and well’ (78.6%, Figure 1), followed by ‘had fun with
late effects relating to their cancer and/or treatment (SD 3.1, range 0– their friends’ (74.6%). When compared to normative data from chil-
15). The five most commonly reported late effects were dental prob- dren without special health care needs, childhood cancer survivors
lems (43.4%), fatigue (38.3%), problems relating to immunity (37.7%), had significantly lower parent-reported overall HRQoL (48.4 vs. 50.6,
memory and learning problems (33.7%) and emotional difficulties p < .009).23 Comparison across items in the Kidscreen-10 within our
(30.3%). Parents commonly reported that they were ‘a little worried’ sample using the Cochran’s Q test indicated that parents were signifi-
or ‘somewhat worried’ about their child having a cancer recurrence cantly more likely to report that their child had problems with feeling
(63.2%) or late effects (64.9%), and 23.6% and 25.3% reported they lonely and feeling sad compared to other items (Figure 1).
were worried ‘a lot’ or ‘a great deal’ about cancer recurrence or late
effects, respectively. Parents had a mean resilience score of 8.56 (range
2–10, SD 1.5). The majority of parents reported that their child cur- 3.2 Parent HRQoL
rently attended a long-term follow-up clinic (78.5%) for survivorship
care. Parents reported that they were ‘satisfied’ (42.9%) or ‘very sat- Survivors’ parents’ average HRQoL was 0.90 (SD 0.12, range 0.14–
isfied’ (37.3%) with the long-term follow-up care they received. 1.00) and 38.5% of parents reported a difference from perfect health.25
Parents commonly indicated problems relating to anxiety/depression
(43.3%), followed by pain (42.3%; Figure 2). On average, parents
3.1 Parent-reported child HRQoL reported their overall health today as 80.1 out of 100 (SD 15.0,
range 30–100). Parents’ overall HRQoL was no different to Australian
On average, parents reported their child’s global HRQoL was 48.4 (SD normative data on both the index score (population mean = 0.91,
10.8, range 27.3–87.9). Parents indicated that their child ‘always’ or p = .475) and overall health rating (population mean = 78.6,
‘very often’ had high HRQoL in most domains, providing the highest p = .163). When considering each domain of HRQoL, the proportion of
6 of 10 FARDELL ET AL .

FIGURE 2 Parents of childhood cancer health-related quality of life (HRQoL) domains compared to population norms. *Significantly different
at p < .05

survivors’ parents experiencing problems did not differ from Australian 3.4 Determinants of parent HRQoL
norms on pain/discomfort (p = .570). Significantly more survivors’ par-
ents reported experiencing problems with anxiety/depression (43.4%) Lower parent HRQoL was significantly associated with lower child
compared to Australian norms (24.7%, p < .0001). Survivors’ parents HRQoL (p = .015), greater fear that cancer would recur in their child
were less likely to report problems with mobility (p < .0001), self-care (p = .001) and lower levels of parent resilience (p = .015; Table 2).
(p = .024), and usual activities (p = .005) when compared to the general No other factors were significant, including age, sex, diagnosis, treat-
population. ment(s), number of late effects experienced, or time since diagnosis.
Given psychological functioning was a particularly vulnerable area
of HRQoL for parents of survivors, in post hoc analyses we sought to
3.3 Determinants of child HRQoL determine factors associated with ratings of problems with anxiety and
depression. The following factors were significantly associated with
We examined the impact that parent and child factors had on overall more anxiety and depression problems in the final logistic multivariable
child HRQoL. In multiple linear regression (Table 2), receiving treat- model: child’s diagnosis of brain cancer (p = .014, OR 22.101, 95% CI:
ments other than surgery (i.e., chemotherapy, radiotherapy, transplant, 1.861–262.514), and lower parent resilience (p < .0001, OR 0.542, 95%
p = .015) and experiencing more late effects (p = .003) were sig- CI: 0.391–0.750). Overall, lower child HRQoL as measured by parent
nificantly associated with worse parent-reported child HRQoL. No report on the Kidscreen-10 was marginally associated with increased
other factors were significantly associated with parent-reported child likelihood of parents reporting problems with anxiety and depression
HRQoL. (p = .053, OR 0.960, 95% CI: 0.921–1.000). No other factors were asso-
In post hoc analyses, we sought to determine factors associated ciated with likelihood of parents reporting problems with anxiety and
with parents rating their child as feeling sad and feeling lonely on depression.
the Kidscreen-10. In the multivariable analysis, only lower parent
resilience was associated with a greater likelihood of their child experi-
encing sadness (p = .039, OR = 0.757, 95% CI: 0.581–0.986). Parents 4 DISCUSSION
were more likely to report their child experienced loneliness if they
had a parent income >$60,000 AUD p.a. (p = .024, OR 1.001, 95% CI: In our study, parents reported that their child’s overall HRQoL was
1.000–1.003) or lower parent resilience (p = .036, OR 0.759, 95% CI: worse than normative data. When individual domains of child HRQoL
0.587–0.982). were considered, parents reported that their child survivor was more
FARDELL ET AL . 7 of 10

TA B L E 2 Associations with child and parent HRQoL

Child HRQoL (Kidscreen-10 T-score) Parent HRQoL (EQ-5D-5L index)

Multiple 95% CI 95% CI Multiple 95% CI 95% CI
Factor Univariate p regression p Beta lower higher Univariate p regression p Beta lower upper
Child factors
Child age .481 .930
Child sex .186 *
.176 −2.279 −5.593 1.036 .686
Diagnosis (overall) coded .485 .224
for ICCC
Surgery .047* .015 −4.209 −7.603 −0.816 .117* .329 .020 −0.020 0.060
Chemotherapy .004* .067 8.737 −0.621 18.095 .341
Radiotherapy .24 .250
BMT .615 .673
Time since diagnosis .728 .795
Number of health issues <.0001 *
.003 −.999 −1.647 −0.351 .241
LTFU attendance .947 .092* .062 −.043 −0.089 0.002
LTFU satisfaction .510 .282
Child HRQoL n/a – – – – .009* .012 .002 0.001 0.004
(Kidscreen-10 T)
Parent factors
Parent sex .356 .464
Education .551 .605
Marital status .917 .970
Place of residence .889 .081* .488 .016 −0.030 0.063
Employment .43 .043 *
.112 −.040 −0.090 0.010
Income .451 .216
Resilience .002* .073 1.060 −0.102 2.223 .000* .013 .017 0.004 0.031
Worried about cancer .72 .001 *
.001 −.026 −0.041 −0.011
recurrence
Worried about late .013* .862 −.133 −1.638 1.373 .007a
effects
Parent HRQoL .009* .103 10.908 −2.246 24.063 n/a – – – –
(EQ-5D-5L index)

Abbreviations: HRQoL, health-related quality of life; ICCC, International Classification of Childhood Cancer; n/a, not applicable.
a
Excluded due to high correlation with worries about cancer.
*
Entered into multiple linear regression.

likely to experience sadness and loneliness compared to all other areas Similar to existing literature,9 our results show psychosocial func-
of functioning except feeling full of energy. Parents were more likely tioning as reported by parents of young survivors may be an area
to report that their child had poor HRQoL if they had received treat- of ongoing vulnerability years after treatment completion. Approxi-
ments other than surgery and had more health issues (late effects) after mately 18% of parents reported that their child experienced prob-
treatment. Lower parent resilience was related to likelihood of parent- lems with feeling sad and feeling lonely. Consistent with others,15,17
reported child sadness and loneliness. Parents of childhood cancer sur- our results indicate that parents of survivors adapt relatively well to
vivors reported that their own overall HRQoL in survivorship years was their child’s cancer when considering overall HRQoL or overall func-
no different to normative data. However, 43.5% parents reported prob- tioning in survivorship. Yet, similarly parents may also remain psycho-
lems with anxiety and/or depression on the EQ-5D-5L. Parents at risk logically vulnerable long after treatment completion. Qualitative par-
of poor HRQoL were those reporting lower child HRQoL, lower levels ent interviews in other studies complement our results and suggest
of resilience and greater worry about the risk of cancer recurrence in parents and young survivors continue to grapple with the emotional
their child. No treatment or diagnostic factors were consistently asso- upheaval and disruption that occurs during diagnosis and treatment in
ciated with parent HRQoL. survivorship.26
8 of 10 FARDELL ET AL .

We found physical symptoms, as measured by more late effects hood cancer survivors. While fathers may initially adapt faster to
experienced in survivorship, and having received treatments other their child’s life-threatening illness/injury, they may experience worse
than surgery (i.e., chemotherapy, radiotherapy and transplant) were mental health in the long-term when compared to mothers.33 Future
significantly associated with worse parent-reported overall child research recruiting both parents would allow better understanding of
HRQoL.12 These results may be related to the symptom burden and the impact of childhood cancer on the family and parent’s long-term
concomitant medical treatment needs experienced by patients and HRQoL. Furthermore, lower educational attainment is a risk factor for
the associated caregiving burden experienced by parents during sur- poorer HRQoL,34 and the results obtained here may not reflect the
vivorship. For example, parent’s perceptions of their child’s cancer experiences of parents with lower educational attainment during their
severity and interference with life have been found to be signifi- child’s survivorship years.
cantly associated with ratings of HRQoL.27 However, caregiving bur- Our response rate was moderate and lower than published psycho-
den mediated this relationship, with high caregiving burden associated oncology study participation rates.35 Although there were no differ-
with reduced HRQoL.27 Previous research has found that some diag- ences between participating and nonparticipating parents on avail-
noses (i.e., brain cancer) and more intense treatment (severity and fre- able demographic characteristics, nonparticipating parents may have
quency of hospitalisations) are associated with worse parent psycho- been those experiencing greater psychological concerns. We were also
logical functioning.28,29 Whilst we found no association between par- unable to undertake age-stratified comparisons on parent HRQoL.
ent HRQoL and treatment and diagnostic factors in our study, we did Combined with our sample size, the generalizability of our results may
find that having a child diagnosed with brain cancer was significantly be compromised. We relied on parent proxy ratings for child HRQoL,
associated with worse psychological functioning in post hoc analyses. which may be discrepant with child self-ratings. Namely, children tend
Our results reinforce the interrelatedness of child and parent to rate themselves as having better HRQoL than their parents.36 How-
HRQoL. Parents who reported they were generally psychologically ever, parent perceptions of their child’s HRQoL may influence health
resilient also reported better HRQoL for their child and themselves. care engagement and transition to self-management of medical follow-
Similarly, parents who perceive their child’s cancer as more uncer- up as young survivors become adults.15,37 Research suggests parent
tain, and have negative attitudes towards illness, are more likely to proxy ratings and child ratings of HRQoL are similar, especially when
report worse adjustment and HRQoL for themselves and their child.30 the child is younger (as in our sample), and when considering domains
Therefore, parent adjustment and coping with their child’s diagnosis of HRQoL that are readily observable.36,38,39 However, future research
may be critical for determining later psychological functioning for the would benefit from including child-reported HRQoL. A further limita-
whole family,12,16,26,29 potentially more so than diagnosis, treatment, tion is our reliance on a sole informant (i.e., parent), which may influ-
subsequent late effects. Further comprehensive research on parental ence the observed relationship between parent and child HRQoL and
psychological functioning and its influence on their child’s health care the predictors due to common method variance.
engagement and survivorship care is therefore warranted.

5 CONCLUSIONS
4.1 Clinical implications

In our study, parents reported that their child experienced reduced

Our results indicate that young survivors experienced reduced HRQoL
overall HRQoL, while overall parent HRQoL was no different to norms.
compared to norms and a subset of parents experienced HRQoL that
There was some evidence for poor psychosocial functioning among
was clinically meaningfully different from perfect health. Accordingly,
both survivors and parents warranting further investigation. Our study
improving patient and parent HRQoL should be a goal of survivorship
indicates modifiable factors, such as fear of cancer recurrence and
care. Our data indicating potential for ongoing psychological impact
fear of late effects, and parent resilience may play a role in determin-
and no consistent relationship with clinical/demographic factors sup-
ing child and parent HRQoL during survivorship. Models of long-term
port arguments for implementing universal psychosocial screening and
follow-up care emphasise managing late effects and possible second
access to appropriate psychological support for children and parents
cancers through identifying patients at high risk due to the treatments
where needed as part of routine long-term follow-up.14,15 Paediatric
received.40,41 Such risk-based approaches may benefit from taking into
oncology survivorship programmes may benefit from embedded psy-
account the potential for poor HRQoL; however, future research is
chosocial care, yet many clinics lack dedicated psychological support
needed on the best ways to identify young survivors, and their parents,
and screening.31 Focusing on parent resilience, together with address-
at risk of poor HRQoL. Development and evaluation of family-centred
ing unmet psychological and information needs,32 may support parent
and resiliency programmes could ultimately support not just childhood
and child HRQoL during survivorship.
cancer survivors’ but also parents’ adjustment during survivorship.

4.2 Limitations ACKNOWLEDGEMENTS

Joanna Fardell and Christina Signorelli are supported by The Kids’
Our sample consisted of mostly well-educated mothers and our results Cancer Project. Claire Wakefield is supported by a Career Develop-
therefore may not accurately reflect the HRQoL of fathers of child- ment Fellowship from the NHMRC of Australia (APP1143767). The
FARDELL ET AL . 9 of 10

Behavioural Sciences Unit (BSU) is proudly supported by the Kids with 11. Barakat LP, Li Y, Hobbie WL, et al. Health-related quality of life of ado-
Cancer Foundation. The BSU’s survivorship research programme is lescent and young adult survivors of childhood brain tumors. Psychoon-
cology. 2015;24(7):804-811.
funded by the Kids Cancer Alliance, The Kids’ Cancer Project and a
12. Racine NM, Khu M, Reynolds K, Guilcher GMT, Schulte FSM. Quality
Cancer Council NSW Program Grant (PG16-02) with the support of of life in pediatric cancer survivors: contributions of parental distress
the Estate of the Late Harry McPaul. The MCRI is supported by the and psychosocial family risk. Curr Oncol. 2018;25(1):41-48.
Victorian Governments’ Operational Infrastructure Support Program. 13. Warner EL, Kent EE, Trevino KM, Parsons HM, Zebrack BJ, Kirchhoff
AC. Social well-being among adolescents and young adults with can-
These funding bodies did not have any role in the study, nor did they
cer: a systematic review. Cancer. 2016;122(7):1029-1037.
have a role in the writing of the manuscript or the decision to sub- 14. Lown EA, Phillips F, Schwartz LA, Rosenberg AR, Jones B. Psychosocial
mit it for publication. ANZCHOG is supported by the Australian Gov- follow-up in survivorship as a standard of care in pediatric oncology.
ernment, through Cancer Australia’s Support for Cancer Clinical Trials Pediatr Blood Cancer. 2015;62(Suppl 5):S514-S584.
15. Kearney JA, Salley CG, Muriel AC. Standards of psychosocial
programmes.
care for parents of children with cancer. Pediatr Blood Cancer.
2015;62(S5):S632-S683.
CONFLICT OF INTEREST 16. Ljungman L, Cernvall M, Grönqvist H, et al. Long-term positive and
The author declare that there is no conflict of interest to disclose in negative psychological late effects for parents of childhood cancer sur-
relation to this manuscript. vivors: a systematic review. PLoS One. 2014;9(7):e103340.
17. Wakefield CE, Mcloone JK, Butow P, Lenthen K, Cohn RJ. Parental
adjustment to the completion of their child’s cancer treatment. Pediatr
DATA AVAILABILITY STATEMENT Blood Cancer. 2011;56(4):524-531.
The data that support the findings of this study are not publicly avail- 18. Buchbinder DK, Fortier MA, Osann K, et al. Quality of life among par-
able due to privacy or ethical restrictions, and the full dataset is not able ents of adolescent and young adult brain tumor survivors. J Pediatr
Hematol Oncol. 2017;39(8):579-584.
to be released due to ethical restrictions. Requests may be made to the
19. Herdman M, Gudex C, Lloyd A, et al. Development and preliminary
authors. testing of the new five-level version of EQ-5D (EQ-5D-5L). Qual Life
Res. 2011;20(10):1727-1736.
ORCID 20. Mccaffrey N, Kaambwa B, Currow DC, Ratcliffe J. Health-related qual-
ity of life measured using the EQ-5D–5L: South Australian population
Joanna E. Fardell https://orcid.org/0000-0001-7334-3475
norms. Health Qual Life Outcomes. 2016;14(1):133.
Christina Signorelli https://orcid.org/0000-0002-7091-0347 21. Rabin R, Oemar M, Oppe M, Janssen B, Herdman M. EQ-5D-5L User
Jordana McLoone https://orcid.org/0000-0002-2604-9975 Guide. EuroQol Group Executive Office; 2011.
Michael Osborn https://orcid.org/0000-0002-1288-9930 22. Erhart M, Ottova V, Gaspar T, et al. Measuring mental health
and well-being of school-children in 15 European countries using
Antoinette Anazodo https://orcid.org/0000-0002-5495-6062
the KIDSCREEN-10 Index. Int J Public Health. 2009;54(2):160-
166.
REFERENCES 23. Ravens-Sieberer U, Erhart M, Rajmil L, et al. Reliability, construct and
1. Robison LL, Hudson MM. Survivors of childhood and adolescent can- criterion validity of the KIDSCREEN-10 score: a short measure for
cer: life-long risks and responsibilities. Nat Rev Cancer. 2013;14:61. children and adolescents’ well-being and health-related quality of life.
2. Bhakta N, Liu Q, Ness KK, et al. The cumulative burden of surviving Qual Life Res. 2010;19(10):1487-1500.
childhood cancer: an initial report from the St Jude Lifetime Cohort 24. Vaishnavi S, Connor K, Davidson JRT. An abbreviated version of the
Study (SJLIFE). Lancet. 2017;390(10112):2569-2582. Connor–Davidson Resilience Scale (CD-RISC), the CD-RISC2: psycho-
3. Geenen M, Cardous-Ubbink M, Kremer LCM, et al. Medical assess- metric properties and applications in psychopharmacological trials.
ment of adverse health outcomes in long-term survivors of childhood Psychiatry Res. 2007;152(2-3):293-297.
cancer. JAMA. 2007;297(24):2705-2715. 25. Pickard AS, Neary MP, Cella D. Estimation of minimally important dif-
4. Henderson TO, Oeffinger KC. Paediatrics: addressing the health bur- ferences in EQ-5D utility and VAS scores in cancer. Health Qual Life
den of childhood cancer survivors - improvements are needed. Nat Rev Outcomes. 2007;5(1):70.
Clin Oncol. 2018;15(3):137-138. 26. Ljungman L, Boger M, Ander M, et al. Impressions that last: particu-
5. Ferrans CE, Zerwic JJ, Wilbur JE, Larson JL. Conceptual model of larly negative and positive experiences reported by parents five years
health-related quality of life. J Nurs Scholarsh. 2005;37(4):336-342. after the end of a child’s successful cancer treatment or death. PLoS
6. Wallander JL, Varni JW. Effects of pediatric chronic physical dis- One. 2016;11(6):e0157076.
orders on child and family adjustment. J Child Psychol Psychiatry. 27. Salvador Á, Crespo C, Martins AR, Santos S, Canavarro MC. Par-
1998;39(1):29-46. ents’ perceptions about their child’s illness in pediatric cancer:
7. The EuroQol Group. EuroQol-a new facility for the measurement of links with caregiving burden and quality of life. J Child Fam Stud.
health-related quality of life. Health Policy. 1990;16(3):199-208. 2015;24(4):1129-1140.
8. Fardell JE, Vetsch J, Trahair T, et al. Health-related quality of life of 28. Sultan S, Leclair T, Rondeau É, Burns W, Abate C. A system-
children on treatment for acute lymphoblastic leukemia: a systematic atic review on factors and consequences of parental distress as
review. Pediatr Blood Cancer. 2017;64(9):e26489. related to childhood cancer. Eur J Cancer Care (Engl). 2016;25(4):616-
9. Vetsch J, Wakefield CE, Robertson EG, et al. Health-related quality of 637.
life of survivors of childhood acute lymphoblastic leukemia: a system- 29. Mccarthy MC, Ashley DM, Lee KJ, Anderson VA. Predictors of acute
atic review. Qual Life Res. 2018;27(6):1431-1443. and posttraumatic stress symptoms in parents following their child’s
10. Yağc-Küpeli B, Akyüz C, Küpeli S, Büyükpamukçu M. Health-related cancer diagnosis. J Trauma Stress. 2012;25(5):558-566.
quality of life in pediatric cancer survivors: a multifactorial assessment 30. Mullins LL, Cushing CC, Suorsa KI, et al. Parent illness appraisals, par-
including parental factors. J Pediatr Hematol Oncol. 2012;34(3):194- ent adjustment, and parent-reported child quality of life in pediatric
199. cancer. Pediatr Hematol Oncol. 2016;33(5):314-326.
10 of 10 FARDELL ET AL .

31. Signorelli C, Wakefield CE, Mcloone JK, et al. Models of childhood 39. Janse AJ, Sinnema G, Uiterwaal CSPM, Kimpen JLL, Gemke RJBJ.
cancer survivorship care in Australia and New Zealand: strengths and Quality of life in chronic illness: children, parents and paediatricians
challenges. Asia Pac J Clin Oncol. 2017;13(6):407-415. have different, but stable perceptions. Acta Paediatr. 2008;97(8):1118-
32. Vetsch J, Fardell JE, Wakefield CE, et al. Forewarned and forearmed”: 1124.
long-term childhood cancer survivors’ and parents’ information needs 40. Frobisher C, Glaser A, Levitt GA, et al. Risk stratification of child-
and implications for survivorship models of care. Patient Educ Couns. hood cancer survivors necessary for evidence-based clinical long-term
2017;100(2):355-363. follow-up. Br J Cancer. 2017;117(11):1723.
33. Darling SJ, Hearps SJC, Muscara F, et al. Psychological trajectories 41. Signorelli C, Wakefield CE, Johnston KA, et al. Re-engage’ pilot
of mothers and fathers following their child’s diagnosis of a life- study protocol: a nurse-led eHealth intervention to re-engage, edu-
threatening illness or injury: a longitudinal investigation. J Clin Psychol. cate and empower childhood cancer survivors. BMJ Open. 2018;8(4):
2019;75(10):1930-1942. e022269.
34. Szende A, Janssen B, Cabases J. Self-Reported Population Health: An
International Perspective Based on EQ-5D. Springer; 2014.
35. Wakefield CE, Fardell JE, Doolan EL, et al. Participation in psychoso-
cial oncology and quality-of-life research: a systematic review. Lancet SUPPORTING INFORMATION
Oncol. 2017;18(3):e153-e165. Additional supporting information may be found in the online version
36. Schulte F, Wurz A, Reynolds K, Strother D, Dewey D. Quality of life in of the article at the publisher’s website.
survivors of pediatric cancer and their siblings: the consensus between
parent-proxy and self-reports. Pediatr Blood Cancer. 2016;63(4):677-
683.
37. Heath G, Farre A, Shaw K. Parenting a child with chronic illness as they
How to cite this article: Fardell JE, Wakefield CE, De Abreu
transition into adulthood: a systematic review and thematic synthesis
of parents’ experiences. Patient Educ Couns. 2017;100(1):76-92. Lourenco R, et al. Long-term health-related quality of life in
38. Yoo HJ, Ra YS, Park HJ, et al. Agreement between pediatric brain young childhood cancer survivors and their parents. Pediatr
tumor patients and parent proxy reports regarding the pediatric func- Blood Cancer. 2021;68:e29398.
tional assessment of cancer therapy-childhood brain tumor survivors
https://doi.org/10.1002/pbc.29398
questionnaire, version 2. Cancer. 2010;116(15):3674-3682.