Parent illness appraisals, parent adjustment, and parent-reported child quality of life in pediatric cancer

Article (PDF Available)inPediatric Hematology and Oncology 33(5):1-13 · July 2016with 158 Reads
DOI: 10.1080/08880018.2016.1198443
Abstract
Psychosocial distress is a salient construct experienced by families of children with newly diagnosed cancer, but little is known about parental appraisal of the child's illness and the subsequent impact this may have on child and parent functioning. The goal of the present study was to examine the interrelationships among multiple parent illness appraisals, parent adjustment outcomes, and parent-reported child quality of life in parents of children diagnosed with cancer. Parents completed measures of illness appraisal (illness uncertainty and attitude toward illness), parent adjustment (general distress, posttraumatic stress, parenting stress), and child quality of life (general and cancer-related). Path analysis revealed direct effects for parent illness uncertainty and illness attitudes on all 3 measures of parent adjustment. Illness uncertainty, but not illness attitudes, demonstrated a direct effect on parent-reported child general quality of life; parenting stress had direct effects on general and cancer-related quality of life. Exploratory analyses indicated that parent illness uncertainty and illness attitudes conferred indirect effects on parent-reported general and cancer-related quality of life through parenting stress. Negative parent illness appraisals appear to have adverse impacts on parents' psychosocial functioning and have implications for the well-being of their child with cancer.
Author content
All content in this area was uploaded by John M. Chaney Regents Professor - Oklahoma State
PEDIATRIC HEMATOLOGY AND ONCOLOGY
http://dx.doi.org/./..
Parent illness appraisals, parent adjustment,
and parent-reported child quality of life in pediatric cancer
Larry L. Mullinsa, Christopher C. Cushingb,KristinaI.Suorsa
a,AlaynaP.Tackett
a, Elizabeth
S. Molzona,SunnyeMayes
c, Rene McNall-Knappc, Alexandria J. Mullinsa,Kaitlyn
L. Gamwella, and John M. Chaneya
aDepartment of Psychology, Oklahoma State University, Stillwater, Oklahoma, USA; bDepartment of Psychology,
University of Kansas, Lawrence, Kansas, USA; cDepartment of Pediatrics, University of Oklahoma Health Sciences
Center, Oklahoma City, Oklahoma, USA
ARTICLE HISTORY
Received  April 
Revised  May 
Accepted  June 
KEYWORDS
Attitude towards illness;
illness uncertainty; oncology;
pediatric cancer;
posttraumatic stress; quality
of life
ABSTRACT
Psychosocial distress is a salient construct experienced by families of
children with newly diagnosed cancer, but little is known about parental
appraisal of the child’s illness and the subsequent impact this may have
on child and parent functioning. The goal of the present study was to
examine the interrelationships among multiple parent illness appraisals,
parent adjustment outcomes, and parent-reported child quality of
life in parents of children diagnosed with cancer. Parents completed
measures of illness appraisal (illness uncertainty and attitude toward
illness), parent adjustment (general distress, posttraumatic stress, par-
enting stress), and child quality of life (general and cancer-related). Path
analysis revealed direct eects for parent illness uncertainty and illness
attitudes on all 3 measures of parent adjustment. Illness uncertainty, but
not illness attitudes, demonstrated a direct eect on parent-reported
child general quality of life; parenting stress had direct eects on gen-
eral and cancer-related quality of life. Exploratory analyses indicated
that parent illness uncertainty and illness attitudes conferred indirect
eects on parent-reported general and cancer-related quality of life
through parenting stress. Negative parent illness appraisals appear to
have adverse impacts on parents’ psychosocial functioning and have
implications for the well-being of their child with cancer.
Introduction
Research on families of children diagnosed with cancer indicates that the majority of parents
and youth will adjust to the demands placed on them by their disease (e.g., Hampel et al. [1]).
However, despite ndings indicating that psychosocial impairment in parents and children
with cancer often mirror those in the general population, a signicant minority continue to
face adjustment diculties in terms of general psychological distress, posttraumatic stress
symptoms,andparentingstress[2–6].
In addition, health-related quality of life (HRQOL) is an area of major concern for children
and adolescents with cancer. Studies indicate that a small and consistent subset of youth with
cancer will experience signicantly decreased HRQOL compared with typically developing
CONTACT Larry L. Mullins, PhD larry.mullins@okstate.edu Department of Psychology, Oklahoma State University, 
North Murray, Stillwater, OK , USA.
©  Taylor & FrancisGroup, LLC
Downloaded by [Oklahoma State University], [John Chaney] at 09:40 21 July 2016
2 L. L. MULLINS ET AL.
healthy children [7].Itisalsoimportanttopointoutthatwhereaschild-reportedHRQOL
appearscentraltounderstandingchildfunctioningduringtheacutetreatmentphasesofill-
ness [8], parent-rated child HRQOL may be more predictive of long-term outcomes for both
caregivers and children [9]. This is likely related to the observation that parent perceptions
of child functioning (particularly HRQOL) are instrumental in ongoing decisions regarding
their child’s medical treatment, regimen management, and utilization of health care resources
overthecourseofillness[10].
Because one of the goals of interdisciplinary oncology programs is returning a family to
normal functioning while minimizing the impact of illness on any given individual, it would
seem crucial to understand the key drivers of child HRQOL to promote healthy adjustment
for all family members throughout the course of treatment. Although considerable research
documents that children’s cognitive appraisals and adjustment are related to child HRQOL,
far fewer studies have examined parent cognitive appraisals and parent adjustment variables
associated with youth HRQOL (e.g., [11–13]). Given empirical evidence pointing to the neg-
ative downstream eects of parent adjustment on child functioning (e.g., [14]), identifying
the key multiple determinants of parent adjustment (e.g., parent illness appraisals) that may
also impact child HRQOL outcomes could prove benecial because they represent potential
targets for psychosocial intervention [15,16].
Parent illness appraisals and parent adjustment links
Illness uncertainty is one particular cognitive appraisal mechanism that has been reliably
associated with negative adjustment outcomes in a number of pediatric chronic illnesses,
including pediatric cancer, e.g., [17,18]. Illness uncertainty has been described as a cogni-
tive appraisal elicited in situations where the meaning of illness-related events are unclear or
unpredictable (e.g., ambiguous illness symptoms, complex treatments, unknown or limited
illness information, episodic illness course [19–21]). Across multiple disease groups, uncer-
tainty has been reliably linked to various adjustment outcomes in both parents and youth
(e.g., [22]). Importantly, parent illness uncertainty has been associated with posttraumatic
stress symptoms in parents of children surviving a brain tumor [23].
Attitude toward illness represents a related, yet distinct, cognitive appraisal stress-
processing mechanism linked to adjustment outcomes. Austin and Huberty [24] describe atti-
tude toward illness as either a positive or negative evaluation of one’s illness that develops as a
function of ongoing experiences with illness treatment, symptoms, and exacerbations, as well
as physical, social, and academic challenges. To date, the majority of investigations indicat-
ing that more negative attitudes are associated with both increased illness severity and poorer
childadjustmentoutcomeshavefocusedontheroleofyouthperceptionsoftheirownillness
(e.g., [24–27]). Of particular relevance to the present study are ndings demonstrating that
parents’ negative perceptions about their child’s i l ln e ss a re a s s oci at e d with increased general
parent distress and parenting-specic stress [28,29].
The present study
Although previous investigations have identied several parent illness appraisal parent
adjustment HRQOL relationships in isolation, no known study has evaluated these con-
structs simultaneously using multiple indices of parent functioning and parent-rated child
qualityoflifeinpediatriccancer.Basedonpreviousliteratureandcontemporarytransactional
stress and coping models of adjustment [30], we conceptualized 3 categories of variables for
Downloaded by [Oklahoma State University], [John Chaney] at 09:40 21 July 2016
PEDIATRIC HEMATOLOGY AND ONCOLOGY 3
Figure . Conceptual diagram. PTSS =posttraumatic stress symptoms; QOL =quality of life.
investigation: (1) parent illness appraisals (illness uncertainty and attitude toward illness; (2)
parent adjustment (parent distress, posttraumatic stress, and parenting stress); and (3) parent
perceptions of child HRQOL (both general and cancer-specic). Specically, we examined
parent illness appraisal mechanisms as primary variables in the development of untoward
parentadjustmentoutcomes,whichinturnserveasmoreproximalvariablesinthedevel-
opment of lower perceptions of child HRQOL. See Figure 1 for our conceptual model and
hypothesized relationships.
It was anticipated that more negative parent illness appraisals would have direct eects
on both poorer parent adjustment and less positive perceptions of child HRQOL. We also
anticipated that increased general parent distress, posttraumatic stress, and parenting stress
would be directly related to less positive perceptions of child HRQOL. Due to insucient
literature establishing the temporal precedence of the variables in the proposed model, we
refrained from hypothesizing specic indirect paths among our variables. However, we did
test all possible indirect paths as a strategy for generating hypotheses for future longitudinal
work examining determinants of both positive and negative family outcomes in the context
of pediatric cancer.
Methods
Participants and procedures
Participants were recruited from a pediatric oncology clinic between August 2009 and
September 2013 in a teaching hospital in the Midwest region of the United States. Eligible care-
givers were identied through the hematology/oncology clinic patient database, and medical
eligibility was assessed through consultation with the attending physician. Eligibility criteria
were (1) parents of children aged 1 to 18 years with pediatric cancer; (2) English was the pri-
mary language for the family; (3) child was not receiving palliative care; and (4) the child was
Downloaded by [Oklahoma State University], [John Chaney] at 09:40 21 July 2016
4 L. L. MULLINS ET AL.
Tab le . Caregiver (N=) demographics.
Demographic Parent (%)
Caregiver status
Mother .
Fath er .
Custodial caregiver .
Missing .
Gender
Male .
Missing .
Ethnicity
Caucasian .
Native American .
African American .
Hispanic .
Asian .
Multiracial .
Other .
Missing .
Marital status
Married .
Never married .
Divorced .
Cohabiting/living with partner .
Other .
Missing .
Household Income
$, .
$,–, .
$,–, .
$,–, .
$,–, .
$, .
on an active treatment protocol or being actively monitored as part of treatment follow-up in
clinic. The hospital’s institutional review board approved the current study.
Eligible caregivers (N=162), self-identied as the legal guardian, were approached dur-
ing their child’s outpatient appointment to participate in a study designed to understand how
families respond to the experience of having a child with cancer. One hundred and thirty-
eight caregivers (85%) agreed to participate and completed measures in a private location
within the clinic. Parents (77% mothers) completed measures of demographic and medi-
cal information, illness uncertainty, attitude toward illness, parenting stress, posttraumatic
stress symptoms, general distress, and both parent-reported general and cancer-specic child
QOL.
Children (53% male) were diagnosed with leukemia/lymphoma (41.0%), non-central ner-
vous system (CNS) solid tumors (26.6%), brain tumors (13.7%), and other cancers (18.7%).
Approximately half the children (53%) were less than 7 years of age (M=8.01; SD =5.22).
Virtually all (97%) were within 2 years of their diagnosis (M=7.80 months; SD =7.40).
Participants were compensated $10 for their participation (see Table 1 for additional sample
demographics).
Measures
Participants completed a demographic questionnaire. Participants also provided information
regarding their child’s cancer diagnosis. All information was conrmed by a member of the
research sta.
Downloaded by [Oklahoma State University], [John Chaney] at 09:40 21 July 2016
PEDIATRIC HEMATOLOGY AND ONCOLOGY 5
To assess parent perceptions of illness-related uncertainty, the Parent Perception of
Uncertainty Scale (PPUS) [31] was utilized. The PPUS asks parents to respond to 31 items
regarding the uncertainty of their child’s illness and treatment (e.g., “I am unsure if my childs
illness is getting better or worse”). Responses are summed to calculate a total score, with higher
values reecting greater perceived illness uncertainty (M=70.34; SD =16.24). The reliabil-
ity and validity for the PPUS are acceptable [31]. Cronbach’s alpha for the current study was
.91.
The Parent Attitude toward Child’s Illness Scale (PATIS) [28] was used to examine parental
perceptions of their child’s illness. The PATIS is a 13-item self-report measure adapted from
the Child Attitude Toward Illness Scale [24] that assesses how parents feel about their child’s
health condition. The content of the items on the parent version were based on the original
child version but contained modied wording directed towards parent illness attitudes. For
this study, the measure was modied to reect parent attitudes specic to their child’s cancer
(e.g., “How fair is it that your child has cancer?”). Higher total scores indicate more posi-
tive attitudes toward the child’s health condition (M=2.59; SD =0.58). In previous studies,
the PATIS has demonstrated good internal reliability (α=.84) and accounts for signicant
variance in parental distress (e.g., [28]). In the present sample Cronbach’s alpha was .82.
The Parenting Stress Index—Short Form (PSI-SF) [32] was utilized to measure general
parenting stress. The PSI-SF is a 36-item self-report measure that assesses stress pertaining
specically to the parent’s relationship with their child. Higher scores indicate greater stress
related to parenting (M=70.78; SD =20.78). Previous research has shown the PSI-SF to have
good reliability and validity (e.g., [33]). Internal consistency in the present sample was high
(α=.91).
The Impact of Events Scale—Revised (IES-R) [34] was utilized to assess symptoms of post-
traumatic stress. Consistent with the authors’ recommendations, instructions were tailored
so that parents responded to the 22 items in the context of their child’s diagnosis of cancer.
Subscale scores assessing intrusive thoughts, hyperarousal, and avoidance were combined to
calculate a total score. Higher scores indicate greater levels of distress (M=27.15; SD =17.98).
Previous research has shown the IES-R to have good reliability and validity [34]. Additionally,
the IES-R has been used widely to measure trauma-related distress in parents of children with
cancer [35–37]. Cronbach’s alpha for the current study was .93.
The Brief Symptom Inventory (BSI) [38] is a 53-item self-report instrument designed to
examine general psychological distress acrossavarietyofsymptomdomains(e.g.,anxiety,
depression,hostility).Forthecurrentstudy,theGlobalSeverityIndexwasusedasthemeasure
of general psychological distress, with higher scores indicating greater psychological distress
(M=0.70; SD =0.64). Research has found the BSI to have good reliability and validity [38].
Cronbach’s alpha in the present sample was .97.
The Pediatric Quality of Life Inventory, General Module (PedsQL-G) [7] is a 23-item instru-
ment used to examine caregiver report of general health-related quality of life in children and
adolescents. Individual items (e.g., “in the past ONE month, how much of a problem has
your child had with worrying about what will happen to him or her”) are reverse-scored,
and linear transformations produce a 0–100 scale. Higher scores indicate better quality of life
(M=64.45; SD =20.50). Research has demonstrated the PedsQL-G to have acceptable relia-
bility and validity within the pediatric cancer population [7].Cronbachsalphainthissample
was .90.
The Pediatric Quality of Life Inventory, Cancer Module (PedsQL-C) [7] is a 27-item instru-
ment used to examine caregiver report of cancer-specic health-related quality of life in chil-
dren and adolescents. Similar to the PedsQL-G, individual items (e.g., “in the past ONE
Downloaded by [Oklahoma State University], [John Chaney] at 09:40 21 July 2016
6 L. L. MULLINS ET AL.
month, how much of a problem has your child had with getting anxious about having blood
drawn”) are reverse-scored and transformed, resulting in a 0–100 scale. Higher scores indicate
betterqualityoflife(M=68.13; SD =15.37). Acceptable reliability and validity have been
demonstrated in previous studies [7]. In the present sample α=.91.
Data analysis
Data were analyzed using Mplus 7.1 [39]. All exogenous constructs were allowed to freely cor-
relate with each other. Child age and parental gender were included as covariates. Model t
wasevaluatedusingthechi-squarestatisticaswellasabsolute(i.e.,rootmeansquareerrorof
approximation [RMSEA]) and relative t statistics (comparative t index [CFI]; non-normed
t index [NNFI]). A nonsignicant chi-square was considered to be the best estimate of model
t, whereas CFI and NNFI thresholds for acceptable t were set at .90 and RMSEA was set
at .08. All paths were standardized on a 1.0–1.0 scale and were considered signicant at
P<.05. Using a model pruning procedure (e.g., [40]), a just identied model was initially
specied (i.e., estimating all 12 hypothesized direct eects) and nonsignicant paths were
constrained to zero until each remaining path was signicant. However, covariates (child
age, parental gender) were not constrained regardless of signicance. The nal pruned model
demonstrated close t to the data (χ2(8, N=138) =8.63, P=.37, RMSEA =.00–.105, CFI
=1.00, NNFI =.99). The modeled eects explained 38% and 31% of the variability in the
exogenous parent-reported general HRQOL and cancer-specic HRQOL variables, respec-
tively. For the exploratory analyses, we conducted a path analysis with 5000 bootstrapped
samples for estimating indirect eects.
Results
Preliminary analyses
A series of preliminary analyses revealed no signicant dierences on any of the modeled
variables across child gender or race. Nonsignicant mean comparisons were also observed
for children with tumors versus those with leukemia/lymphoma. Finally, time since diagnosis
was uncorrelated with any of the modeled variables.
Primary analyses of direct eects
Signicant direct eects were observed between both illness-specic cognitive appraisal
variables (illness uncertainty [PPUS]; parent attitude toward illness [PATIS]) and the 3 adjust-
ment outcome variables (parent general distress [BSI], parenting stress [PSI], and post-
traumatic stress symptomology [IES-R]). Specically, one illness-specic cognitive appraisal
variables, illness uncertainty (PPUS), was positively associated all 3 adjustment outcome
variables (parent general distress [BSI], β=.38; parenting stress [PSI-SF], β=.42; and
posttraumatic stress symptomology [IES-R], β=.35). The parent attitude toward the child’s
illness (PATIS) was negatively associated with parent general distress (BSI, β=−.28), parent-
ing stress (PSI-SF, β=−.25), and posttraumatic stress symptomology (IES-R, β=−.43). All
6 of these eects were in the anticipated direction. Combined, illness uncertainty (PPUS) and
theparentattitudetowardthechildsillness(PATIS)explained30%ofthevariabilityingeneral
distress (BSI), 42% in posttraumatic stress symptomology (IES-R), and 32% in parenting stress
(PSI-SF; see Figure 2).
Downloaded by [Oklahoma State University], [John Chaney] at 09:40 21 July 2016
PEDIATRIC HEMATOLOGY AND ONCOLOGY 7
Figure . Final model. PTSS =posttraumatic stress symptoms; QOL =quality of life.
Results related to parent-reported child health-related quality of life (HRQOL) were mixed.
Illness uncertainty (PPUS), but not the parents attitude toward the childs illness (PATIS),
was signicantly related to health-related quality of life-general (PedsQL-G, β=−.29). Fur-
ther, parenting stress (PSI-SF) was the only adjustment variable signicantly associated with
health-related quality of life—general (PedsQL-G, β=−.32). Little support was found for the
direct inuence of parent illness appraisals (illness uncertainty or parent attitude toward the
child’s illness) or adjustment (parent distress, parenting stress, or posttraumatic stress sympto-
mology) on cancer-related quality of life (HRQOL-CS, PedsQL-C). The parent attitude toward
the child’s illness (PATIS) was not associated with cancer-related quality of life (PedsQL-C).
However, a signicant relationship between illness uncertainty (PPUS) and was observed
with cancer-related quality of life (PedsQL-C, β=−.29).Oftheadjustmentvariables,only
parenting stress (PSI-SF) demonstrated an association with the cancer-related quality of life
(PedsQL-C, β=−.29); no eects were observed for parent distress (BSI) or posttraumatic
stress symptomology (IES-R) on cancer-related quality of life (PedsQL-C).
Exploratory analyses of indirect eects
To generate hypotheses for future studies, illness appraisaladjustmentHRQOL indirect
eects were examined. Results revealed a signicant illness uncertainty (PPUS) parenting
stress (PSI-SF) general quality of life (PedsQL-G) indirect path (95% condence interval
[CI] =−.22 to .06), indicating that increased parent illness uncertainty inuenced parent
estimates of child general quality of life indirectly through increased parenting stress. Parent-
ing stress also carried a signicant indirect eect in the parent attitude toward the child’s illness
(PATIS) parenting stress (PSI-SF) general quality of life (PedsQL-G) path (95% CI =.02
Downloaded by [Oklahoma State University], [John Chaney] at 09:40 21 July 2016
8 L. L. MULLINS ET AL.
to .14). Because no direct association was observed between the parent attitude toward the
child’s illness (PATIS) and general quality of life (PedsQL-G), exploratory results may suggest
that the eect of parent attitude toward illness on child general quality of life was exclusively
indirect, through increased parenting stress.
Results also indicated that both the illness uncertainty (PPUS) parenting stress (PSI-SF)
cancer-related quality of life (PedsQL-C) and the parentsattitudetowardthechildsillness
(PATIS) parenting stress (PSI-SF) cancer-related quality of life (PedsQL-C) indirect paths
were signicant (95% CI =−.22 to .04 and 95% CI =.01 to .14, respectively). Similar to
theexploratoryresultsforgeneralqualityoflife,theabsenceofdirecteectsforeitherillness
uncertainty (PPUS) or the parents’ attitude toward the child’s illness (PATIS) on cancer-related
quality of life (PedsQL-C) suggests that the signicant impacts of both parent illness appraisal
measures on cancer-related quality of life were due to their indirect inuence on elevated levels
of parenting stress.
Discussion
The purpose of the current study was to examine the complex, multiple interrelation-
ships among parent illness appraisals, parental adjustment outcomes, and parent-perceived
HRQOL constructs simultaneously in a single path model. In this manner, we sought
to better understand key factors that drive outcomes in families facing pediatric cancer.
Results partially supported our conceptual framework. Specically, both illness-specic cog-
nitive appraisal variables (illness uncertainty and illness attitudes) demonstrated signicant
associations with each of the 3 parent adjustment outcomes (general parent distress, post-
traumatic stress symptoms, and parenting stress). Results were more mixed with regard to
parent-reported child health-related quality of life. Parent illness uncertainty, but not attitude
toward illness, was associated with children’s general health-related quality of life; neither ill-
ness appraisal variables were related to cancer-specic health-related quality of life. Of the 3
parent adjustment indices, only parenting stress was signicantly associated with child health-
related quality of life (both general and cancer-related).
Our exploratory analyses cautiously suggest that examining indirect eects may be needed
to fully describe the linkages between parent illness appraisals and parent perceptions of
child health-related quality of life. For example, both parent illness uncertainty and parent
illness attitudes were indirectly related to parent perceptions of general and cancer-related
health-related quality of life through their inuence on parenting stress. In general, this
exploratory nding provides cautious optimism for our conceptual parent illness appraisal
parent adjustment parent-reported quality of life path model. Importantly, parenting stress
emerged as a pivotal intermediary parent adjustment variable linking parent illness appraisals
toqualityoflifeoutcomes.
The observed associations among parent illness uncertainty, parent attitude toward illness,
and all 3 parent adjustment measures in our model were consistent with the pediatric chronic
illness literature (e.g., [16,41]). However, it is noteworthy that both illness uncertainty and ill-
ness attitudes made unique contributions to explaining each of these multiple indices of par-
ent adjustment simultaneously. Thus, despite the signicant associations observed between
illness uncertainty and illness attitudes, both variables were independently related to general
parent distress, posttraumatic stress symptoms, and parenting stress. Further, these ndings
are particularly intriguing in the context of additional results observed in the path analysis.
To illustrate, despite the multiple independent appraisal adjustment links, only parenting
Downloaded by [Oklahoma State University], [John Chaney] at 09:40 21 July 2016
PEDIATRIC HEMATOLOGY AND ONCOLOGY 9
stress exerted direct eects on parent-reported child quality of life outcomes. In other words,
both negative illness appraisals were associated with the parent adjustment outcomes that one
might intuitively associate with pediatric cancer (e.g., general parent distress and posttrau-
matic stress [36,42]). However, neither of these adjustment outcomes explained additional
variance in parents’ general or cancer-related perceptions of child quality of life above that
accounted for by general parenting stress.
The absence of direct or indirect associations for general parent distress and posttraumatic
stress symptoms with either of the HRQOL indices may shed some light on the nature of the
parent adjustment constructs and parent-reported HRQOL examined in our study. Although
no direct tests were performed, examination of the βsinFigure 2 indicate that whereas general
parent distress and posttraumatic stress measures were highly associated, parenting-specic
stress and general parent distress shared substantially less variance by comparison. More-
over, parenting stress was unrelated to posttraumatic stress. It may be that parent adjustment
variables such as general parent distress and posttraumatic stress capture parents’ emotional
struggles in dealing with a child’s illness and aremorecloselyassociatedwithemotion-specic
domainsofchildfunctioning(e.g.,anxiety,depression[43]). Although health-related quality
of life measures include child emotional adjustment items, they also assess a broad spectrum
of child functioning domains (e.g., school, physical). Thus, the direct relation between parent-
ing stress and parents’ perceptions of health-related quality of life may suggest that the added
stress from illness demands superimposed on routine parenting (e.g., household tasks, family
schedules)maybemorelikelytoinuenceparent-perceivedchildhealth-relatedqualityof
life relative to more emotion-focused parent adjustment outcomes. This nding might also
provide some insight into why better agreement is observed for parent-child ratings across
the functional domains of child health-related quality of life than for emotional and social
domains (e.g., [44]).
Clinical implications
Our data suggest that when parents experience their child’s illness as more uncertain and/or
hold more negative attitudes toward illness, multiple adverse eects on parent adjustment may
result. With regard to increased illness uncertainty in particular, direct reductions in both
parent adjustment and parent-perceived child health-related quality of life may be potential
outcomes. Although speculative, results of the exploratory analyses suggest that both illness
uncertainty and illness attitudes promote elevated levels of parenting stress, which in turn
leads to decreases in parent-perceived health-related quality of life—perhaps reecting par-
ents’ internal dialogue about their child’s health (e.g., “Everything about my child’s illness is
up in the air” [uncertainty] and/or “I hate that that my child has cancer [negative attitude]
“I feel cut o from my friends, spouse, other children” [parenting stress] “My child doesn’t
have a chance to be like other kids” [quality of life]).
Clinically, our results suggest that early screening of parents and children at risk for
developing psychosocial diculties may prove benecial for targeting interventions aimed
at illness-related cognitive appraisals (e.g., [45]). Both illness uncertainty as well as attitudes
toward illness represent potentially malleable mechanisms that t well into existing cognitive-
behavioral clinical frameworks. Our previous research has demonstrated that interventions
utilizing illness-specic parent appraisals (i.e., illness uncertainty) as an organizing frame-
work are eective in attenuating parental distress in parents of children with type 1 diabetes
(e.g., [15]) and, importantly, in parents of children with newly diagnosed cancer ([46, 47]).
Downloaded by [Oklahoma State University], [John Chaney] at 09:40 21 July 2016
10 L. L. MULLINS ET AL.
Limitations
The present results should be interpreted in light of several important methodological con-
siderations. First, the current sample represented a group of children who were currently on
treatment or being actively followed up in clinic. As such, the results cannot be generalized
to samples of youth who are further out from time of diagnosis or who are o treatment with
no clinical contacts (see Vrijmoet-Wiersma et al. [48]). However, the rst 2 to 3 years fol-
lowing diagnosis (the period best represented by our current sample) is a critical period for
parents, with early adjustment being associated with subsequent adjustment outcomes (e.g.,
[22,49]). It is also important to note that our sample was heterogeneous in terms of diag-
noses. Although we found no dierences between broad subtypes of disease, illness severity
mayhavecontributedtothendings.Futureresearchwillthusneedtocarefullyexaminethe
possible role of disease status.
Further, our data were gathered exclusively by self-report method from single informants.
Because we utilized parents’ ratings of child HRQOL as the primary dependent measure
rather than children’s ratings, it could be argued that parents who endorsed both nega-
tive attitudes regarding their child’s illness and their own personal adjustment may have
also been characterized by pessimistic views generally, including negative perceptions of
theirchildsqualityoflife(seeHood[50]). However, evidence suggests that the mental
health status of parents with a chronically ill child does not inuence ratings of their child’s
health-related quality of life (e.g., [51]). Moreover, if method and/or informant variance
hadbeenprimarilyresponsibleforourndings,wewouldhaveexpectedoneof2out-
comes: either (1) wholesale direct associations with quality of life would have emerged for
both illness appraisal variables and all 3 parent adjustment dimensions or (2) signicant
overlapping variance among the variables would have obscured most (if not all) indepen-
dent associations between the variables. Neither of these outcomes was observed. Neverthe-
less,eventhoughparent-reportedchildHRQOLplaysanimportantroleinmanyaspects
of disease management [10],itshouldnotbeconsideredasubstituteforchildself-reported
HRQOL. Future studies would benet from utilizing both sources of information to delineate
more precisely the complex interrelationships among parent and child outcomes in pediatric
cancer.
Finally, despite being informed by contemporary theory, we realize our model does not
comprise an exhaustive list of parent cognitive and/or adjustment variables that contribute to
the variability in child HRQOL outcomes. Moreover, although our sequential path model
tsthedatawell,thecross-sectionalnatureofourstudycannottellusthetruetempo-
ralnatureoftherelationsamongthevariablesunderstudy(see[52]). Longitudinal stud-
ies are needed to examine prospectively whether parents’ varying levels of illness appraisals,
adjustment, and their perceptions of child quality of life toward the beginning of their
child’s cancer journey can predict dierential patterns of parent and child outcomes over
time.
In summary, the current study illustrates the complexity of the parent illness appraisal
parent adjustmentparent-reported child quality of life links in the context of family
outcomes in pediatric cancer. To our knowledge, this is one of the rst studies to use a path
analysis approach to examine these interrelationships simultaneously with parents across
multiple domains, in an attempt to identify additional salient targets of intervention (e.g., atti-
tudes toward illness, parenting stress) related to both parent and child outcomes. Hopefully,
such results can better inform the development of eective, evidence-based interventions for
families of children with cancer (e.g., [53]).
Downloaded by [Oklahoma State University], [John Chaney] at 09:40 21 July 2016
PEDIATRIC HEMATOLOGY AND ONCOLOGY 11
Acknowledgments
We would like to thank the families and sta at the Jimmy Everest Center for Cancer and Blood Disor-
ders in Children for their gracious participation in this project.
Declaration of interest
The authors report no conicts of interest. The authors alone are responsible for the content and writing
of the paper.
References
[1] Hampel P, Rudolph H, Stachow R, Lab-Lentzsch A, Petermann F. Coping among children and
adolescents with chronic illness. Anxiety Stress Coping. 2005;18:145–155.
[2] Barakat LP, Kazak AE, Meadows AT, Casey R, Meeske K, Stuber ML. Families surviving childhood
cancer: a comparison of posttraumatic stress symptoms with families of healthy children. JPediatr
Psychol. 1997;22:843–859.
[3] Hung JW, Wu YH, Yeh CH. Comparing stress levels of parents of children with cancer and parents
of children with physical disabilities. Psychooncology. 2004;13:898–903.
[4] McCarthy MC, Ashley DM, Lee KJ, Anderson VA. Predictors of acute and posttraumatic stress
symptoms in parents following their child’s cancer diagnosis. JTraumaStress. 2012;25:558–
566.
[5] Pai AL, Greenley RN, Lewandowski A, Drotar D, Youngstrom E, Peterson CC. A meta-analytic
review of the inuence of pediatric cancer on parent and family functioning. JFamPsychol.
2007;21:407–415.
[6] Phipps S, Long A, Hudson M, Rai SN. Symptoms of post-traumatic stress in children with
cancer and their parents: eects of informant and time from diagnosis. Pediatr Blood Cancer.
2005;45:952–959.
[7] Varni JW, Burwinkle TM, Katz ER, Meeske K, Dickinson P. The PedsQL in pediatric cancer: relia-
bility and validity of the Pediatric Quality of Life Inventory Generic Core Scales, Multidimensional
Fatigue Scale, and Cancer Module. Cancer. 2002;94:2090–2106.
[8] Eiser C, Vance YH, Horne B, Glaser A, Galvin H. The value of the PedsQLTM in accessing quality
of life in survivors of childhood cancer. Child Care Health Dev. 2003;29:95–102.
[9] Kazak AE, Barakat L. Brief report: Parenting stress and quality of life during treatment for child-
hood leukemia predicts child and parent adjustment after treatment ends. JPediatrPsychol.
1997;22:749–758.
[10] Varni JW, Limbers CA, Burwinkle TM. Parent proxy-report of their children’s health- related
quality of life: an analysis of 13,878 parents’ reliability and validity across age subgroups using the
PedsQL 4.0 Generic Core Scales. Health Qual Life Outcomes. 2007;5:xx–xx.
[11] Herzer M, Denson LA, Baldassano RN, Hommel KA. Patient and parent psychosocial factors
associated with health-related quality of life in pediatric inammatory bowel disease. JPediatr
Gastroenterol Nutr. 2011;52:295.
[12] Hullmann SE, Wolfe-Christensen C, Meyer WH, McNall-Knapp RY, Mullins LL. The rela-
tionship between parental overprotection and health-related quality of life in pediatric
cancer: the mediating role of perceived child vulnerability. Qual Life Res. 2010;19:1373–
1380.
[13] Fortier MA, Batista ML, Wahi A, Kain A, Strom S, Sender LS. Illness uncertainty and quality of
life in children with cancer. JPediatrHematolOncol. 2013;35:366–370.
[14] Ryan J, Ramsey RR, Fedele D, Mullins LL, Jarvis J, Chaney JM. A longitudinal examination of the
parent—child distress relationship in children with juvenile rheumatic diseases. Rehabil Psychol.
2010;55:286–291.
[15] Ho A, Mullins LL, Gillaspy S, Van Pelt J, Page M, Chaney JM. An intervention to decrease uncer-
tainty and distress among parents of children newly diagnosed with diabetes: a pilot study. Fam
Syst Health. 2005;23:329–342.
Downloaded by [Oklahoma State University], [John Chaney] at 09:40 21 July 2016
12 L. L. MULLINS ET AL.
[16] Mullins LL, Wolfe-Christensen C, Pai ALH, et al. The relationship of parental overprotection,
perceived child vulnerability, and parenting stress to uncertainty in youth with chronic illness. J
Pediatr Psychol. 2007;32:973–982.
[17] Grootenhuis MA, Last BF. Predictors of parental emotional adjustment to childhood cancer. Psy-
chooncology. 1997;6:115–128.
[18] Grootenhuis MA, Last BF. Adjustment and coping by parents of children with cancer: a review of
the literature. Support Care Cancer. 1997;5:466–484.
[19] Mishel MH. Perceived uncertainty and stress in illness. Res Nurs Health. 1984;7:163–171.
[20] Mishel MH. Reconceptualization of the uncertainty in illness theory. JNursScholarsh.
1990;22:256–262.
[21] Neville KL. Uncertainty in illness: an integrative review. Orthop Nurs. 2003;22:206–214.
[22] Fedele DA, Mullins LL, Wolfe-Christensen C, Carpentier MY. Longitudinal assessment of mater-
nal parenting capacity variables and child adjustment outcomes in pediatric cancer. JPediatHema-
tol Oncol. 2011;33:199–202.
[23] Fuemmeler BF, Mullins LL, Marx BP. Posttraumatic stress and general distress among parents of
childrensurvivingabraintumor.Child Health Care. 2001;30:169–182.
[24] Austin JK, Huberty TJ. Development of the Child Attitude toward Illness Scale. JPediatrPsychol.
1993;18:467–480.
[25] Heimlich TE, Westbrook LE, Austin JK, Cramer JA, Devinsky O. Brief report: Adolescents’ atti-
tudestowardepilepsy:furthervalidationoftheChildAttitudeTowardIllnessScale(CATIS).J
Pediatr Psychol. 2000;25:339–345.
[26] LeBovidge JS, Lavigne JV, Miller ML. Adjustment to chronic arthritis of childhood: the roles of
illness-related stress and attitude toward illness. JPediatrPsychol. 2005;30:273–286.
[27] Ramsey RR, Bonner M, Ryan J, Mullins LL, Chaney JM. A prospective examination of attitudes
toward illness and depressive symptoms in youth with juvenile rheumatic diseases. JDevPhys
Disabil. 2013;25:171–180.
[28] Ryan JL, Mullins LL, Ramsey RR, et al. Caregiver demand and parent distress in juvenile rheumatic
diseases: parent attitude toward illness as a mediator. JClinPsycholMedSettings. 2013;20:351–360.
[29] Svavarsdóttir EK, Rayens MK. American and Icelandic parents’ perceptions of the health status of
their young children with chronic asthma. JNursScholarsh. 2003;35:351–358.
[30] Thompson RJ, Gustafson KE. Adaptation to Chronic Childhood Illness.Washington,DC:Ameri-
can Psychological Association; 1996.
[31] Mishel MH. Parents’ perception of uncertainty concerning their hospitalized child. Nurs Res Pract.
1983;22:256–262.
[32] Abidin RR. Parenting Stress Index (PSI) Manual. 3 ed. Charlottesville, VA: Pediatric Psychology
Press; 1995.
[33] Hasket ME, Ahern LS, Ward CS, Allaire JC. Factor structure and validity of the Parenting Stress
Index—Short Form. JClinChildPsychol. 2010;35:302–312.
[34] Weiss DS, Marmar CR. The Impact of Events Scale—Revised. In: xxx, eds. Assessing Psychological
Trauma and PTSD; New York, NY: Guildford Press; 1997:399–411.
[35] Dunn MJ, Rodriguez EM, Barnwell AS, et al. Posttraumatic stress symptoms in parents of children
with cancer within six months of diagnosis. Health Psychol. 2012;31:176.
[36] Kazak AE, Boeving CA, Alderfer MA, Hwang W, Reilly A. Posttraumatic stress symptoms during
treatment in parents of children with cancer. JClinOncol. 2005;23:7405–7410.
[37] Nornberg AL, Boman KK. Mothers and fathers of children with cancer: loss of control during
treatment and posttraumatic stress at later follow-up. Psychooncology. 2013;22:324–329.
[38] Derogatis LR, Spencer PM. The Brief Symptom Inventory (BSI): Administration, Scoring and Pro-
cedures Manual 1. Baltimore, MD: Clinical Research; 1982.
[39] Muthén L, Muthén B. MplusUsersGuide. 7th ed. Los Angeles, CA: Muthén & Muthén; 1998–
2012.
[40] Kline RB. Principles and Practice of Structural Equation Modeling. New York, NY: Guilford Press;
2011.
[41] Cousino M, Hazen R. Parenting stress among caregivers of children with chronic illness: a sys-
tematic review. JPediatrPsychol. 2013;38:809–829.
Downloaded by [Oklahoma State University], [John Chaney] at 09:40 21 July 2016
PEDIATRIC HEMATOLOGY AND ONCOLOGY 13
[42] Jurbergs N, Long A, Ticonia L, Phipps S. Symptoms of posttraumatic stress in parents of children
with cancer: are they elevated relative to parents of healthy children? JPediatrPsychol. 2009;34:4–
13.
[43] Morris A, Gabert-Quillen C, Delahanty D. The association between parent PTSD/depression
symptoms and child PTSD symptoms: a meta-analysis. JPediatrPsychol. 2012;37:1076–1088.
[44] Eiser C, Morse R. Can parents rate their child’s health-related quality of life? Results from a sys-
tematic review. Qual Life Res. 2001;10:347–357.
[45] Kazak AE, Prusak A, McSherry M, et al. The Psychosocial Assessment Tool (PAT): pilot data on
a brief screening instrument for identifying high risk families in pediatric oncology. Fam Syst
Health. 2001;19:303–317.
[46] Mullins LL, Fedele DA, Chan M, et al. A clinic-based interdisciplinary intervention for mothers
of children newly diagnosed with cancer: a pilot study. JPediatrPsychol. 2012;37:1104–1115.
[47] Fedele DA, Hullmann SE, Chan M, et al. Impact of parent-based interdisciplinary interven-
tion for mothers on adjustment in childnren newly diagnoses with cancer. JPediatrPsychol.
2013;38:531–540.
[48] Vrijmoet-Wiersma C, Hoekstra-Weebers J, Margreet de Pender WW. Psychometric qualities of
the Dutch version of the Pediatric Inventory for Parents (PIP): a multi-center study. Psychooncol-
ogy. 2010;19:368–375.
[49] Steele RG, Dryer ML, Phipps S. Patterns of maternal distress among children with cancer and their
association with child emotional and somatic distress. JPediatrPsychol. 2004;29:507–517.
[50] Hood K. The inuence of caregiver depressive symptoms on proxy report of youth depressive
symptoms: a test of the depression-distortion hypothesis in pediatric type 1 diabetes. JPediatr
Psychol. 2009;34:294–303.
[51] Ferro M, Avison W, Campbell M, Speechley K. Do depressive symptoms aect mothers’ reports
of child outcomes in children with new-onset epilepsy? Qual Life Res. 2010;19:955–964.
[52] Maxwell SE, Cole DA. Bias in cross-sectional analyses of longitudinal mediation. Psychol Methods.
2007;12:23.
[53] Law EF, Fisher E, Fales J, Noel M, Eccleston C. Systematic review and meta-analysis of parent
and family-based interventions for children and adolescents with chronic medical conditions. J
Pediatr Psychol. 2014;39:866–886.
Downloaded by [Oklahoma State University], [John Chaney] at 09:40 21 July 2016
  • Article
    Objective: Caregivers of pediatric cancer patients are at risk for posttraumatic stress symptoms (PTSS). Previous literature has identified caregiver illness uncertainty as a predictor of PTSS, yet little is known about the mechanism by which illness uncertainty may affect PTSS. Rumination, or perseverations about the cause and consequences of an event, has been related to posttraumtic stress disorder in other populations. However, limited research of this relationship exists for pediatric cancer caregivers. Further, no studies have evaluated rumination in relation to illness uncertainty. The current study examined rumination in relation to illness uncertainty and PTSS in caregivers with children actively receiving treatment for cancer. It was hypothesized that rumination would be related to illness uncertainty and would mediate the relationship between illness uncertainty and PTSS. Methods: Caregivers (N = 59) completed the Parent Perception of Uncertainty Scale, Ruminative Responses Scale, and the Impact of Event Scale-Revised. Results: illness uncertainty was identified as a significant predictor of rumination (B = 0.148, p = .015), and rumination was a significant predictor of PTSS (B = 1.83, p < .001). Biased-corrected bootstrap regression analysis revealed that rumination mediated the relationship between illness uncertainty and PTSS (R2 = 0.53, p < .001). Conclusions: Rumination was identified as a mediator between the relationship of illness uncertainty and PTSS. These findings underscore the importance of rumination in caregivers of children with cancer, as a potential construct for identifying caregivers at risk of PTSS, and as a possible mechanism for targeted intervention.
  • Article
    Illness blogs are the online narrative expression of the experience of illness and its treatment. The purpose of the present research was to explore, describe, and analyze blog narratives created by parents during their child’s cancer experience in the hope that knowledge generated would amplify the voices of these vulnerable families. The study aimed to answer this question: What themes are evident in illness blogs created by a parent when a child has cancer? The purposive sample of 14 parent blogs included publicly accessible, English language narratives that contained descriptions of life with a child who had undergone treatment for acute lymphocytic leukemia (ALL; five blogs analyzed) or neuroblastoma (nine blogs analyzed) in the previous 5 years or who was currently undergoing treatment for these types of cancer. Analysis resulted in discovery of new knowledge of the uncertainty inherent in daily family life during illness and treatment. The parents’ vivid depictions of the quest for balance while living with prolonged uncertainty during the illness experience suggested new ways to understand experiences of parents of children with cancer.
  • Article
    The purpose of this article is to report an integrative review of uncertainty in the illness experience, describe the uncertainty in illness theory, and to discuss the importance of assessing the appraisal of uncertainty experienced in illness with individuals and families faced with illness.
  • Article
    Full-text available
    Objective To quantify the effects of parent- and family-based psychological therapies for youth with common chronic medical conditions on parent and family outcomes (primary aim) and child outcomes (secondary aim). Methods MEDLINE, EMBASE, and PsycINFO were searched from inception to April 2013. 37 randomized controlled trials were included. Quality of the evidence was evaluated using GRADE criteria. Data were extracted on parent, family, and child outcomes. Results Pooled psychological therapies had a positive effect on parent behavior at posttreatment and follow-up; no significant improvement was observed for other outcome domains. Problem-solving therapy (PST) improved parent mental health and parent behavior at posttreatment and follow-up. There was insufficient evidence to evaluate cognitive-behavioral and systems therapies for many outcome domains. Conclusions Parent- and family-based psychological therapies can improve parent outcomes, with PST emerging as particularly promising. Future research should incorporate consensus statements for outcomes assessment, multisite recruitment, and active comparator conditions.
  • Article
    Full-text available
    Objective To critically review, analyze, and synthesize the literature on parenting stress among caregivers of children with asthma, cancer, cystic fibrosis, diabetes, epilepsy, juvenile rheumatoid arthritis, and/or sickle cell disease. Method PsychInfo, MEDLINE, and Cumulative Index to Nursing and Allied Health Literature were searched according to inclusion criteria. Meta-analysis of 13 studies and qualitative analysis of 96 studies was conducted. Results Caregivers of children with chronic illness reported significantly greater general parenting stress than caregivers of healthy children (d =. 40; p = ≤.0001). Qualitative analysis revealed that greater general parenting stress was associated with greater parental responsibility for treatment management and was unrelated to illness duration and severity across illness populations. Greater parenting stress was associated with poorer psychological adjustment in caregivers and children with chronic illness. Conclusion Parenting stress is an important target for future intervention. General and illness-specific measures of parenting stress should be used in future studies. © 2013 The Author [email protected] /* */
  • Article
    The present study was aimed to investigate age and gender effects on coping with everyday stressors among children and adolescents with chronic illness and healthy controls. Patients (8–13 years of age) with asthma (n=47), atopic dermatitis (n=52), and cancer (n=57) were compared to healthy controls (n=158) matched by age, gender, and grade. Self-report data on coping with academic and interpersonal stressors were collected. The primary results indicated that coping with everyday stressors was improved in children and adolescents with chronic illness compared to healthy controls. Thus, patients reported less passive avoidance on cross-situational coping and tended to show more situation-specific coping with social and school-related stressors than healthy controls. Additionally, among the clinical groups, patients with atopic dermatitis and cancer scored higher on positive self-instructions than patients with asthma. Conclusively, the results suggest that coping with a chronic illness may lead to more effective coping with everyday stressors. Implications for evaluating coping styles in patients with chronic illness are discussed.
  • Article
    Full-text available
    Background: Illness uncertainty is prevalent in children with cancer and has been associated with increased psychological distress. The relationship between illness uncertainty and quality of life in pediatric cancer patients remains unclear. The aim of the present study was to examine illness uncertainty as a predictor of health-related quality of life in children diagnosed with cancer. It was hypothesized that child-reported illness uncertainty would be negatively associated with child health-related quality of life. Procedure: Children aged 8 to 18 years old and receiving treatment for cancer were recruited to participate in this study. One hundred twenty children and their parent(s) completed measures of illness uncertainty, pain, anxiety, and quality of life during a routine visit to the Cancer Center at Children's Hospital of Orange County. Results: Illness uncertainty was significantly associated with child age (P=0.02), overall health-related (P<0.001) and cancer-related (P<0.001) quality of life, but not with treatment status (on/off chemotherapy) or demographic variables including sex and household income. Regression analyses statistically controlling for age, anxiety, and pain revealed that illness uncertainty significantly predicted child-reported cancer-related and health-related quality of life (P<0.01) as well as parent-reported cancer-specific quality of life (P<0.01). Conclusions: Illness uncertainty is prevalent and associated with lower quality of life in children diagnosed with cancer. Improved communication with children regarding disease state, treatment expectations, and prognosis may alleviate uncertainty and improve functioning in this vulnerable patient population.
  • Article
    Full-text available
    Parents of youth with juvenile rheumatic diseases (JRD) often take on illness management responsibilities that can become burdensome, potentially resulting in poor parent adjustment outcomes. However, not all caregivers will experience increased distress as a result of variability in stress appraisals. The current study examined the role of parent illness attitudes in the relation between perceived caregiver demand and parental distress. Youth (N = 70) ages 7-18 years diagnosed with a JRD and their parents were recruited from a pediatric rheumatology clinic. Parents completed measures of caregiver demand, parental distress, and illness attitudes. Hierarchical regression revealed a relationship between caregiver demand and parental distress. A significant relationship was also found between caregiver demand and parent illness attitudes, as well as parent illness attitudes and parental distress. Thus, parent illness attitudes mediated the relationship between caregiver demand and parental distress. Techniques aimed at altering negative illness attitudes may help parents cope with their caregiving responsibilities.
  • Article
    Objective: To determine if maternal distress predicts child adjustment outcomes or if child adjustment outcomes predict maternal distress among children newly diagnosed with cancer, and if a parent-focused intervention has downstream effects on child adjustment. Methods: Mothers (n = 52) were randomly assigned to a clinic-based, interdisciplinary intervention for parents of children newly diagnosed with cancer. Measures of maternal distress and child adjustment were collected at baseline, posttreatment, and follow-up. Results: A lagged relationship was identified between maternal distress and child internalizing symptoms, but not externalizing symptoms. The parent intervention reduced child internalizing and externalizing symptoms at follow-up. Only the child internalizing symptoms effect was mediated by reduced maternal distress. The child externalizing symptoms effect was mediated by unobserved parent factors. Conclusions: This study provides support for illness adjustment and coping models that emphasize the role of parent factors in driving child adjustment outcomes and is encouraging for future parent-focused intervention research.