Journal of Pediatric Psychology, Vol. 28, No. 1, 2003, pp. 5-15
© 2003 Society of Pediatric Psychology
Comparing Parental Distress, Family Functioning, and the Role of Social Support for Caregivers With and Without a Child With Juvenile Rheumatoid Arthritis
Children's Hospital Medical Center and the University of Cincinnati
All correspondence should be sent to Cynthia A. Gerhardt, Division of Hematology/Oncology, Children's Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, Ohio 45229-3039. E-mail: cindy.gerhardt{at}chmcc.org.
 |
Abstract |
|---|
 Top Abstract IntroductionMethodResultsDiscussionReferences |
|---|
Objective To assess parental distress, family functioning, and social support among parents of children with a lifetime diagnosis of juvenile rheumatoid arthritis (JRA) and comparison families. Methods Parents of 64 children with JRA (64 mothers, 46 fathers) completed questionnaires and in-home interviews along with 64 matched comparison families. Average time since diagnosis for children with JRA was 70 months. Results Families of children with JRA generally reported levels of parental distress, family functioning, and social support similar to those for comparison families. More mothers of children with JRA exceeded the clinical cutoff on the SCL-90-R than comparison mothers. Although disease characteristics and social support did not distinguish subgroups of parents at greater risk for problems, family supportiveness and conflict were associated with caseness for mothers of children with JRA. Conclusions Families of children with JRA exhibited substantial resilience over the long term. Further multisite study of children recently diagnosed and with more severe forms of JRA is warranted to determine intervention needs, especially for mothers.
Key words: parents; families; adjustment; juvenile rheumatoid arthritis; social support.
|
Introduction |
|---|
|
TopAbstractIntroductionMethodResultsDiscussionReferences |
|---|
Affecting 30,000 to 50,000 children in the United States each year (Lawrence et al., 1998
),
juvenile rheumatoid arthritis (JRA) is the most common pediatric rheumatic
disease and one of the most common chronic illnesses in childhood
(Cassidy, 1997). JRA is a
heterogeneous group of conditions with considerable variability in the
severity and chronicity of symptoms. Although some children may have multiple
remissions of their disease, JRA can cause long-term physical disability
(Peterson, Mason, Nelson, O'Fallon, &
Gabriel, 1997), and up to two thirds of children may have active
disease in adulthood (Minden et al.,
2000; Peterson et al.,
1997; Zak & Pedersen,
2000). Children with JRA can experience acute and chronic pain in
inflamed joints, decreased mobility due to contractures and joint
deterioration, vision problems, and growth retardation. Systemic involvement
can cause organ damage (e.g., heart or liver) and, on rare occasions, can
result in death. Treatment is palliative, not curative. It typically includes
restrictions on activities and frequent medications that can cause
uncomfortable side effects.
From a systems perspective, the diagnosis of a childhood chronic illness
such as JRA has the potential to disrupt parental and family functioning
(Kazak, 1989). Parents must
manage their own feelings of uncertainty and loss of control, as they face the
added responsibilities and daily hassles associated with caring for a child
with special needs (Quittner, Opipari,
Regoli, Jacobsen, & Eigen, 1992;
Quittner et al., 1998). It is
not uncommon to see evidence of mild to moderate levels of problematic
adjustment for parents of children with a chronic illness
(Jessop, Reissman, & Stein,
1988; Silver, Bauman, &
Ireys, 1995). Mothers of chronically ill children are particularly
at risk, presumably because they assume the greatest responsibility for the
day-to-day care and medical needs of their child
(Noll et al., 1995;
Silver et al., 1995).
Limited work has examined the specific impact of JRA on parental distress
and family functioning, with conflicting results. A recent review concluded
that families of children with JRA may experience multiple difficulties due to
the social, emotional, and financial impact of the disease
(Reisine, 1995). For example,
Vandvik, Hoyeraal, and Fagertun
(1989) found that over two
thirds of parents of children with JRA reported moderate to severe family
difficulties (e.g., parental health problems, family conflict, and lack of
social support), and more than half reported recent stressful life events.
However, this study did not have a control group. As with other chronic
pediatric conditions, mothers of children with JRA are presumed to be at
greater risk for psychological distress than fathers
(Lustig, Ireys, Sills, & Walsh,
1996; Timko, Stovel, &
Moos, 1992; Vandvik &
Eckblad, 1991). Concern for the well-being of these mothers has
prompted randomized clinical trials to decrease their isolation and distress
(Ireys, Sills, Kolodner, & Walsh,
1996).
Other data show that JRA has no significant detrimental impact on parents
and families (e.g., Daltroy et al.,
1992; Myones, Williams,
Billings, & Miller, 1988). For example, in several controlled
studies, families of children with various rheumatic diseases were found to
display levels of adjustment comparable to those of families of healthy
children (Harris, Newcomb, & Gewanter,
1991; Myones et al.,
1988). Although one study indicated that mothers of children with
JRA experienced greater symptoms of depression and general distress than
mothers of healthy children, average scores for these mothers fell within the
normal range of functioning (Frank et al.,
1998). Similarly, Timko et al.
(1992) found that mothers of
children with JRA were more distressed than fathers were, but both groups had
levels of distress within the normal range.
Little is known about what might account for the variability in parental
and family adjustment. Several studies have suggested that disease factors and
social support may influence outcomes (e.g.,
Lustig et al., 1996;
McCormick, Stemmler, & Athreya,
1986; Timko et al.,
1992). With respect to disease factors, families of children with
more serious and debilitating disease appear to be at higher risk for
problematic adjustment and parental strain
(Jessop et al., 1988;
Silver et al., 1995). Specific
to JRA, greater disease severity and functional impairment have been
associated with greater psychological distress among mothers
(Lustig et al., 1996). Greater
functional impairment among children with JRA also has been associated with
increased family stress (McCormick et al.,
1986), increased maternal psychological symptoms
(Lustig et al., 1996), and
poorer concurrent functioning among both mothers and fathers
(Timko et al., 1992). In
contrast, maternal distress has not been associated with disease severity in
one study (Vandvik & Eckblad, 1990), and higher levels of maternal
distress have been found when children exhibited mild disease activity as
compared to none or moderate/severe disease activity
(Daltroy et al., 1992). Daltroy
et al. speculated that mild disease may create difficulties, because it is
less visible and results in less support for children and families.
With respect to social support, previous work has identified two potential
mechanisms for its role in influencing distress
(Cohen & Willis, 1985).
First, social support may have an overall beneficial effect for any
individual, regardless of the number of stressful events to which he or she is
exposed (main effect). Unfortunately, a lack of social support has been
reported by parents of children with JRA
(Ireys et al., 1996;
Vandvik et al., 1989), perhaps
because heightened isolation results from their child's restricted activities
and from the additional time devoted to child care. This lack of social
support and increased isolation may contribute to greater parental distress
(Jessop et al., 1988;
Silver et al., 1995). A
greater availability of social resources has been associated with lower levels
of depression for parents of children with JRA
(Timko et al., 1992), as well
as other chronic conditions (Kronenberger
& Thompson, 1992).
Second, social support may play a more predominant role when individuals
are exposed to particularly stressful circumstances or numbers of stressful
events. This stress by support interaction would result in a stronger
association between support and distress for individuals experiencing higher
levels of stress. Parents of children with JRA may worry about the health of
their child (Gerhardt et al., in
press) and experience numerous stressful life events or daily
hassles (Vandvik et al., 1989,
1991). These stressors may include administering daily medications, clinic
visits, and hospitalizations that would not be expected among parents of
healthy children. Thus, social support and distress may be more strongly
associated for parents of children with JRA than for controls.
The literature on psychosocial morbidity in parents and families of children with JRA presents a number of methodological shortcomings, including the use of unstandardized measures, inappropriate comparison groups, poor recruitment rates, ascertainment bias, and a lack of information from fathers. The purpose of this study was to evaluate parental distress, family functioning, and social support from the perspective of caregivers of children with JRA and comparison caregivers without a chronically ill child. The impact of disease factors also was examined. Based on prior research and clinical observations of the challenges associated with having a child with JRA, the following hypotheses were developed: (a) caregivers of children with JRA will report greater parental distress, greater family difficulties, and fewer social resources than comparison caregivers, with parental distress being more pronounced for mothers of children with JRA than fathers; (b) greater disease severity will be associated with greater parental distress and family difficulties; and (c) parents who report more social support will report less distress (main effect), and this effect will be stronger for parents of children with JRA (interaction).
|
Method |
|---|
|
TopAbstractIntroductionMethodResultsDiscussionReferences |
|---|
Participants and Procedures
Following approval by the institutional review board, rosters of children with a lifetime diagnosis of JRA who were receiving treatment at a large children's hospital were used to identify potential participants. The center is the only facility with board-certified pediatric rheumatologists within a 50-mile catchment area, and a hospital tax levy ensures treatment for disadvantaged youths. The four inclusion criteria for children with JRA were (a) a diagnosis of JRA according to American College of Rheumatology classification standards (Cassidy et al., 1986
), (b) age 8-14 years old, (c) no full-time special education,
and (d) residence within 50 miles of the medical center. Of the 78 eligible
families, 64 participated (4 were not located, 3 refused, and 7 schools
declined participation in the initial phase of the study; see
Noll et al., 2000).
Disease severity for the previous 6 months was rated by the child's
pediatric rheumatologist to overlap with the timing of data collection.
Severity was classified as mild or moderate/severe based on disease onset and
course, the presence of iritis/uveitis, radiographic evaluation,
pharmacotherapy, number of active joints, and Steinbrocker Functional Class
rated by the physician on a scale of 1 to 4 (e.g., completely able to largely
incapacitated; Hochberg et al.,
1992). Any patient receiving methotrexate was considered at least
moderate/severe. Radiologic evidence of moderate/severe disease was defined by
the presence of joint space narrowing or subchondral bone erosion. Thirtyfour
(53%) had mild disease, and 30 (47%) had moderate/severe disease. In addition
to disease severity, the rheumatologist rated each child's disease status as
active (n = 32, 50%) or in full/partial remission (n = 32,
50%). Active disease included the presence of synovitis as defined by swelling
or painful loss of motion. Partial remission was defined as the lack of
objective synovitis but continued use of medication. Disease course was
classified as pauciarticular (n = 31, 48%), polyarticular (n
= 26, 41%), or systemic (n = 7, 11%). Mean time since diagnosis was
69.77 months (SD = 35.89, range = 10-149).
The initial phase of the study involved an assessment of the social
functioning of children with JRA from the perspective of classmates
(Noll et al., 2000).
Subsequently, matched comparison families (COMP) were recruited from lists of
classmates who were the same gender and race as the target child and closest
in age. The family of the classmate whose birthday was closest to the child
with JRA was contacted first and invited to participate in a home assessment.
If they did not wish to participate, the parents of the child with the next
closest birthday were called, and so on, until a comparison family was
recruited. Comparison families were screened to ensure the absence of a
chronic illness in the family. Of these families, 59 (92%) were first choices,
and the remaining families were second or third choices.
Data were collected in the home by two research assistants during a time that was convenient for the family and when the child was not hospitalized (i.e., no acute medical crisis). After providing informed consent/assent, parents and children independently completed several questionnaires. Families received $100 for their participation. Data were collected from 64 mothers in each group (JRA/COMP), 46 fathers of children with JRA (15 families were single mother, 3 fathers declined), and 40 COMP fathers (22 families were single mother, 2 fathers declined). All measures were completed by parents only.
Measures
Demographic Questionnaire. This instrument assesses basic
background characteristics of the respondent, including marital status,
religion, education, occupation, income, and number and age of offspring.
Family socioeconomic status (SES) was computed using the Revised Duncan (TSEI;
Nakao & Treas, 1992). This
occupation-based measure is a contemporary indicator of SES that is sensitive
to changes in occupational attainment
(Hauser, 1994).
Symptom Checklist 90-Revised (SCL-90-R;
Derogatis, 1983). This
90-item self-report inventory measures current psychological symptoms. It
yields nine dimensions of psychological distress and three global indices of
functioning (i.e., Global Severity Index [GSI], Positive Symptom Distress, and
Positive Symptom Total). The GSI, considered the best single summary measure,
combines information on the number and intensity of the symptoms reported.
Positive Symptom Distress is an indicator of the intensity of symptoms
experienced, and Positive Symptom Total represents the number of symptoms
endorsed. This instrument was selected because of its sensitivity to general
distress (Moore, Gilliss, & Martinson,
1988), especially internalizing difficulties. The SCL-90-R
demonstrates adequate internal scale consistency (.77-.90) and reasonably good
test retest reliability (1 week; .78-.90)
(Derogatis, 1983). Previous
work has supported the concurrent validity of the measure
(Brophy, Norvell, & Kiluk,
1988) and the sensitivity of the measure to difficulties
experienced by parents of children with cancer
(Noll et al., 1995). Our
primary analysis of this instrument uses the GSI, but scores and exploratory
analyses are reported for all scales to facilitate comparisons within the
scientific literature.
Family Environment Scale (FES;
Moos & Moos,
1994). The FES is a 90-item questionnaire designed to
assess the family's social climate. Initially, the FES demonstrated adequate
reliability for 10 summary scales, but later work indicated lower
reliabilities (Loveland-Cherry, Youngblut,
& Kline Leidy, 1989; Roosa
& Beals, 1990). For this reason, four higher-order factor
scores (i.e., Family Relationship Index [FRI], Supportive, Conflicted, and
Controlling) were used in this study
(Kronenberger & Thompson,
1990; Moos & Moos,
1994). Although the FRI has some overlap with the Conflicted
scale, it was included for reference due to its use in previous studies.
Internal consistencies for the 10 subscales are adequate (.61-.78) and
test-retest reliabilities are also reasonably good across a 2-month period
(.68-.86; Moos & Moos,
1994). Considerable evidence is presented in the instrument manual
(Moos & Moos, 1994)
supporting the concurrent validity of this measure. The FES has been used
previously in studies of families of children with JRA
(Harris et al., 1991;
Myones et al., 1988).
Norbeck Social Support Interview (NSSI;
Norbeck, Lindsey, & Carrieri,
1981). In an interview format, the respondent is asked to
generate a list of significant others in his or her life (network size) and
then to answer a series of six structured questions that allow for measurement
of the parent's satisfaction with each person nominated. Previous work has
demonstrated good predictive validity, test-retest reliability (r =
.85-.92), and internal consistency (
> .85)
(Norbeck & Anderson, 1989;
Norbeck et al., 1981;
Norbeck, Lindsey, & Carrieri,
1983). Two scores reflecting network size and perceived functional
support are derived from this measure. The NSSI has been used previously with
parents of chronically ill children
(Florian & Krulik, 1991;
Noll et al., 1994).
Analysis Plan
Two-tailed, independent t
tests1 and chi-square
analyses ( = .05) were used as appropriate to compare JRA and COMP
families on background variables. Two-tailed, independent t tests
( = .05) also were used to compare both types of families on parental
distress (GSI), family functioning (FRI, Supportive, Conflicted, and
Controlling), and social support (Network Size and Perceived Functional
Support). Two-tailed, independent t tests were used to examine the
association between multiple disease factors, including severity (mild or
moderate/severe), status (active or full/partial remission), type
(pauciarticular or polyarticular/systemic), and measures of parental distress,
family functioning, and social support. Pearson correlations were used to
examine the relationship between disease duration and parental distress and
family functioning.
Hierarchical multiple regressions were used to investigate the nature of
the association between social support and parental distress (GSI). As
suggested by Aiken and West
(1991), all predictor variables
were centered with respect to the mean score for each variable prior to entry
into the regression equation. A main effect for group status (JRA/COMP) was
first considered followed by entry of one of the two measures of support
(i.e., to examine the main effect of social support). The interaction of group
and support was entered last to test our final hypothesis.
Holm's corrections for multiple procedures was used to reduce Type 1 error
in selected families of analyses (for details, see
Holland & Copenhaver,
1988, or Holm,
1979). This procedure limits Type 1 error probability to at most
, and it allows for increased power compared to traditional
Bonferroni-type corrections. For this reason, corrections were completed for
exploratory analyses using the SCL-90-R subscales that were not hypothesized
effects. All subscale scores were treated as a family of variables separately
for mothers and for fathers to facilitate corrections. Holm's correction was
also applied to the family of tests involving each disease variable (i.e.,
severity, activity, type, duration). This was done due to the large number of
tests completed and our lack of a priori hypotheses for disease activity,
type, and duration.
Using GPOWER for power calculations
(Faul & Erdfelder, 1992),
we found that the sample of 128 mothers produced ample power (.89-.96) to
detect medium effects for t tests (d = .5) and multiple
regressions (f2 = .15). For fathers, the sample of 86
allowed enough power (.74-.84) to detect medium effects for t tests
(d = .5) and multiple regressions (f2 = .15).
These effect sizes are consistent with those reported in the
literature.2
|
Results |
|---|
|
TopAbstractIntroductionMethodResultsDiscussionReferences |
|---|
Background Characteristics
Independent t tests indicated that JRA and COMP families did not differ significantly on a variety of background characteristics, including parent age, SES, parental education, number of children in the home, and child age (Table 1). The modal SES for both groups reflected occupations in clerical or sales positions (e.g., secretaries, sales clerks) or skilled blue collar jobs (e.g., craftsmen, machinists). Marital status did not differ between mothers of children with JRA (77% married) versus comparison mothers (66% married),
2
(4, n = 128) = 4.15, ns.
|
Parental Distress
No significant differences were found on the GSI scores within either group
of mothers or fathers. Additional independent t tests for the
SCL-90-R subscales indicated that mothers of children with JRA reported
significantly higher scores for Positive Symptom Distress than comparison
mothers (Table II). When Holm's
correction was applied, this was no longer significant. Mothers did not differ
on the remaining SCL-90-R subscales. Fathers of children with JRA did not
differ significantly from comparison fathers on the SCL-90-R subscales
(Table III). In addition,
mothers and fathers of children with JRA did not differ significantly from one
another on SCL-90-R scores.
|
|
Further analyses were completed to compare the number of parents in each
group who exceeded the clinical cutoff (i.e., a t score > 63 for
GSI scores or for at least two subscales). Significantly more mothers of
children with JRA (n = 29, 46%) were in the clinical range than
comparison mothers (n = 18, 28%), 2 (1, n =
127) = 4.37, p < .05. For fathers of children with JRA, 22 (48%)
were in the clinical range versus 13 (33%) comparison fathers,
2 (1, n = 85) = 2.08, ns. Disease factors,
background characteristics, and social support did not significantly
distinguish subgroups based on clinical status. However, clinical status for
mothers of children with JRA was significantly associated with lower
Supportive (Mnonclin = 33.93, SD = 5.58,
Mclin = 27.82, SD = 7.79), t (50) =
3.53, p = .001, and higher Conflicted (Mnonclin =
-10.55, SD = 4.16, Mclin = -6.62, SD =
4.93), t (61) = -3.44, p = .001, scores on the FES. FES
scores were not associated with clinical status for COMP.
Family Functioning
Four independent t tests were calculated for the higher-order
factors on the FES to compare family functioning among those with and without
a child with JRA (Tables IV and
V). There were no significant
differences between the two groups based on mother or father report.
|
|
Social Support
Independent t tests compared the size of social support networks
and perceived functional support for JRA and COMP families. There were no
significant differences between the two groups based on mother or father
report (Tables IV and
V). Hierarchical multiple
regressions showed no significant main effects of group status and social
support on parental distress and family functioning. In addition, no
significant interactions between group status and social support were found in
predicting parental distress and family functioning
Disease Factors
Pearson correlations indicated that time since diagnosis was not associated
significantly with parental distress, family functioning, or social support.
Other disease factors were previously collapsed into dichotomous variables due
to the small numbers of children with more severe forms of JRA or inactive
disease. Independent t tests indicated that parental distress, family
functioning, and social support did not differ as a result of mild versus
moderate/severe disease or active versus full/partial remission. Having a
child with polyarticular or systemic disease was associated significantly with
mother and father report of higher GSI scores and with mother report of higher
Positive Symptom Total compared to having a child with pauciarticular disease.
When Holm's correction was applied, these results were no longer
significant.
|
Discussion |
|---|
|
TopAbstractIntroductionMethodResultsDiscussionReferences |
|---|
Previous work has indicated that families of children with JRA may be at risk for psychosocial difficulties (Reisine, 1995
), but data have
been limited and subject to numerous methodological problems. Although a few
controlled studies have painted a brighter picture
(Harris et al., 1991;
Myones et al., 1988), these
have been influenced by small samples and limited power. Poor recruitment and
the use of unstandardized measures have been frequent problems. Also, there is
a lack of information regarding the adjustment of fathers. The culmination of
these methodological shortcomings has resulted in uncertainty regarding the
degree to which these families are at risk for psychosocial difficulties and
in need of intervention. Seeking to ameliorate these problems by using a controlled design, this study assessed the impact on families of having a child with a lifetime diagnosis of JRA. Families of children with JRA were found to exhibit levels of adjustment quite similar to those for comparison parents, with the exception that a greater proportion of mothers of children with JRA met criteria for "caseness" on the SCL-90-R. Disease factors, background characteristics, and social support were not associated with clinical status. However, for mothers of children with JRA, clinical status was associated with Supportive and Conflicted scores on the FES. Contrary to predictions, mothers of children with JRA were not at greater risk for difficulties compared to fathers of children with JRA. Similar levels of social support were reported by parents of children with JRA and COMP parents. No main effects for social support or interactions with group status were identified. Disease factors such as severity, activity, type, and time since diagnosis were not associated significantly with psychosocial variables.
Studies that have found significant problems for families of children with
JRA have tended to be those with the weakest designs and no controls. Often
family disruption has been assessed using unstandardized measures that have
included a variety of problems, such as parental health concerns and a lack of
social support (e.g., Vandvik et al.,
1989). Our finding that families of children with JRA exhibited
functioning similar to that of comparison families is consistent with other
controlled studies using the FES (Harris
et al., 1991; Myones et al.,
1988). However, these studies used small samples. Our sample
yielded enough power to detect medium effects, but a multisite investigation
with a sample of 500-600 would be necessary to detect small effects and
further reduce Type II error.
Although mothers of children with JRA have been presumed to be at greater
risk for difficulties than fathers, both groups often have fallen within the
normal range of functioning (e.g., Timko
et al., 1992). Of studies using adequate controls, only one found
significant differences between mothers of children with JRA and comparison
mothers (Frank et al., 1998).
Specifically, mothers of children with JRA reported greater symptoms of
depression and general distress than controls, but again, these scores fell
well within the normal range of functioning. Furthermore, the average SCL-90-R
depression score (M = 58.08, SD = 10.11, n = 98)
was nearly identical to the sample of mothers in this study (M =
57.72, SD = 10.25). Although we found that mothers of children with
JRA were more likely than comparison mothers to meet criteria for caseness, we
did not find significant differences on any other indices of distress.
The finding regarding caseness on the SCL-90-R was perplexing, because mothers did not differ significantly on the individual subscales. In addition, disease, background, and social support variables failed to demonstrate significant associations with clinical status. Intuitively, it fits that mothers of children with JRA who also were in the clinical range may perceive less family support and more conflict on the FES, but the same was not true for COMP mothers. Furthermore, information about the causal or temporal associations between these variables is unknown, limiting our ability to predict which subgroups of mothers will be at risk for difficulties. Future research can potentially replicate these findings and provide longitudinal data to clarify this issue.
Researchers have emphasized the importance of evaluating the well-being of
fathers and the unique contribution they offer to understanding family
functioning (Phares & Compas,
1992). Unfortunately, psychological research has been notorious
for the exclusion of fathers and for their misuse as comparisons for mothers.
Contrary to our expectations, fathers of children with JRA did not report
greater difficulties for themselves or their families than fathers of healthy
children. In the one study that has reported data from fathers, scores fell
within the normal range of functioning
(Timko et al., 1992). Our
findings further support the notion that having a child with JRA does not
significantly increase risk for paternal distress.
Had we chosen to make comparisons to normative data in lieu of a control
group, we may have concluded that JRA was associated with excessive parental
distress. Some t scores for fathers on the SCL-90-R were nearly one
standard deviation above an expected score of 50, but comparison fathers
reported equally elevated scores. It is striking that approximately half of
the JRA sample and a third of the COMP sample met criteria for caseness. The
rate of caseness in our COMP sample is over three times that expected based on
standardized scoring and available normative information (i.e., t
score > 63, 90th percentile). One might argue that our comparison sample
may have been unusual, but our matching procedure ensured that JRA and COMP
families were nearly identical on multiple demographic and social risk
factors. Comparisons to normative data have been criticized because they are
dependent on the quality of the normative sample and can be susceptible to
regional and cohort effects in spite of considerable effort to obtain a
contemporary and representative normative sample
(Achenbach & Howell, 1993;
Sandberg, Meyer-Bahlburg, & Yager,
1991). Recent national events highlight the caution needed when
using norms for measures of distress.
Our previous work using the SCL-90-R with parents of children with cancer
(Noll et al., 1995) or sickle
cell diseases (Noll et al.,
1994) has shown a similar pattern of elevated scores for both
parents of children with a chronic illness and controls. Despite previous
efforts to obtain more information (sampling procedures, demographics,
recruitment rates, etc.) about the "normative" sample from the
publisher of the SCL-90-R (Noll et al.,
1994), we have not been successful. The SCL-90-R manual was first
published in 1977, and 85% of the nonpatient normal sample was single, despite
an average age of 46 years. Thus, we strongly suggest exercising caution when
using these norms. These findings underscore the importance of including
demographically well-matched controls in future research and displaying
thoughtfulness with the use of normative data. Alternative approaches to
identifying controls for research in pediatric psychology might include
conducting neighborhood searches, random digit dialing, or using a
"snowball" technique (see Noll, Ris, Bukowski, Davies, &
Koontz, 1992).
Contrary to expectations, parent and family functioning did not vary as a
result of disease factors despite the use of a range of objective physician
ratings for disease severity, activity, type, and time since diagnosis.
Although there is conflicting evidence, most research has indicated that more
severe disease and functional impairment may increase risk for psychosocial
difficulties (Lustig et al.,
1996; Silver et al.,
1995). A comprehensive rating of disease severity was developed
for this study by incorporating multiple indicators, including functional
status. However, mixing indicators of both severity and functional status may
have confounded the problem. Different strategies for assessing disease
factors have been employed previously (e.g.,
Lustig et al., 1996;
McCormick et al., 1986), which
has made direct comparison across studies difficult. In addition, studies
often have been confounded by the use of the same source for measures of
psychosocial outcomes and disease factors (i.e., mothers; e.g., Canning,
Heller, Kelleher, 1996; Vandvik et al., 1991). These source variance and
measurement issues (e.g., the lack of a valid, operational measure of
severity) have been difficult to disentangle and may account for our lack of
findings. Last, despite our rigorous recruitment strategies, we had a limited
number of children with more severe diagnoses. Collaboration across multiple
sites may be necessary to include children with a wider variety of disease
characteristics.
Limited data have suggested that some families of children with JRA may
lack social resources and support (Ireys
et al., 1996; Vandvik et al.,
1989), but our study found no differences in social support
network size or perceived functional support for JRA and COMP families. As
with disease severity, investigators have used multiple methods of assessing
social support (e.g., Timko et al.,
1992; Vandvik et al.,
1989), many of which have included single item assessments (e.g.,
"Do you have a confidant?" or "Do you have sufficient
support?"). In addition, no one has evaluated differences in levels of
social support and its impact on distress among parents of children with JRA
and controls. Using an established measure of social support and a controlled
design, we failed to support our hypothesis that families of children with JRA
would have fewer social resources than comparisons or that social support
would be more strongly associated with parental distress in families of
children with JRA. These results are consistent with previous studies using
the same methodology with families of children with cancer
(Noll et al., 1995) and sickle
cell diseases (Noll et al.,
1994).
This research has several methodological weaknesses. One should note that
the sample had a limited number of children with severe disease, which may
have affected the ability to detect problems for some groups of families at
higher risk for difficulties. In addition, data were obtained at only one
point in time at an average of 6 years postdiagnosis, reflecting long-term
adjustment rather than acute reactions. Earlier work has suggested that
parents of children with chronic illness
(Wallander & Varni, 1998),
including JRA (Vandvik et al., 1991), may have more distress around the time
of diagnosis. However, JRA is an unpredictable, chronic disease that can cause
long-term disability (Peterson et al.,
1997). Although many children experience long-term remission, up
to two thirds of children may have recurrent disease in adulthood
(Minden et al., 2000;
Peterson et al., 1997;
Zak & Pedersen, 2000).
Because there are no established criteria for remission and cure for JRA,
adult criteria have been used (Pinals,
Masi, & Larsen, 1981), and a consensus group is currently
studying this issue. Two thirds of our sample had active disease or only
partial remission, which meant that these children were receiving ongoing
treatment at the time of the assessment. Furthermore, our rheumatologists were
reluctant to classify the remaining children as "cured" due to the
chronic, episodic nature of JRA, the relatively young age of the children, and
the significant risk of relapse.
Other methodological points include the fact that data were obtained from only one center and may not accurately reflect the status of parents and families from other locations. In addition, we used one source of information and questionnaires to measure our variables of interest. Observations, multiple reporting sources, Q-sorts, or interviews need to be included. Finally, our sample size limited our ability to detect variables that may moderate the relationship between caring for a child with JRA and parental distress. A longitudinal study across multiple sites that begins shortly after diagnosis is clearly needed.
Overall, this study supports Masten's
(2001) assertion that
resilience is more common than traditionally believed. These families
demonstrated a remarkable ability to adapt to the demands of having a child
with a diagnosis of JRA. Alternatively, it is feasible that parents do not
find having a child with JRA very demanding. This seems less likely, as
limited data available near diagnosis (Vandvik et al., 1991) suggest
considerable distress. The process of how parents adapt and what promotes
these positive outcomes is less clear. Currently, data do not support
broad-based interventions for all families of children with JRA, but some
mothers may warrant additional attention.
|
Acknowledgments |
|---|
This project was supported by a grant to Robert B. Noll from the National Arthritis Foundation.
|
Notes |
|---|
This paper was reviewed and accepted during the term of the previous editor, Anne E. Kazak, PhD, ABPP.
1 Because each child with JRA was matched to a classmate of the same gender,
race, and age, it could be argued that a matched pairs t test would
have been more appropriate. This would have been more powerful than a
between-groups test if the reduction in the error term (a function of
r) outweighed the effect of a loss in degrees of freedom (df
= N - 1 rather than n1 = n2 -
2). Historically, we have found modest correlations on psychosocial variables
between target and comparison families. Rather than reducing error, this
matching procedure appears to equate groups on key demographic factors that
otherwise may have contributed to spurious effects. 
2 An astute reviewer suggested that a better test of the buffering hypothesis
might assess the impact of social support on the association between disease
severity and parental distress. Although we calculated adequate power to
detect a large overall effect for the regression equation, power would have
been extremely limited to adequately test the interaction effect with only a
portion of the sample (i.e., 64 mothers or 45 fathers in the JRA group). Not
surprisingly, when we ran these tests, none of the regressions was
significant.
Received July 16, 2001; revision received December 20, 2001; revision received March 16, 2002; accepted March 20, 2002
|
References |
|---|
|
TopAbstractIntroductionMethodResultsDiscussionReferences |
|---|
Achenbach, T. M., & Howell, C. T. (1993). Are America's children getting worse? A 13-year comparison. Journal of the American Academy of Child and Adolescent Psychiatry, 32, 1145-1154.[Web of Science][Medline]
Aiken, L. S., & West, S. G. (1991). Multiple regression: Testing and interpreting interactions. Newbury Park, CA: Sage.
Brophy, C. J., Norvell, N. K., & Kiluk, D. J. (1988). An examination of the factor structure and convergent and discriminant validity of the SCL-90-R in an outpatient clinic population. Journal of Personality Assessment, 52, 334-340.[CrossRef][Web of Science][Medline]
Canning, R. D., Harris, E. S., & Kelleher, K. J.
(1996). Factors predicting distress among caregivers to children
with chronic medical conditions. Journal of Pediatric
Psychology, 21,
735-749.
Cassidy, J. T. (1997). Rheumatic diseases of childhood. In W. N. Kelley, E. D. Harris, S. Ruddy, & C. B. Sledge (Eds.), Textbook of rheumatology (5th ed., pp. 1207-1224). Philadelphia: W. B. Saunders.
Cassidy, J. T., Levinson, J. E., Bass, J. C., Baum, J., Brewer, E. J., Fink, C. W., Hanson, V., Jacobs, J. C., Masi, A. T., Schaller, J. G., Fries, J. F., McShane, D., & Young, D. (1986). A study of classification criteria for a diagnosis of juvenile rheumatoid arthritis. Arthritis & Rheumatism, 29, 274-281.[Web of Science][Medline]
Cohen, S., & Willis, T. A. (1985). Stress, social support, and the buffering hypothesis. Psychological Bulletin, 98, 310-357.[CrossRef][Web of Science][Medline]
Daltroy, L. H., Larson, M. G., Eaton, H. M., Partridge, A. J.,
Pless, I. B., Rogers, M. P., & Liang, M. H. (1992).
Psychosocial adjustment in juvenile arthritis. Journal of Pediatric
Psychology, 17,
277-289.
Derogatis, L. R. (1983). SCL-90-R administration, scoring, and procedures manual-II. Towson, MD: Clinical Psychometric Research.
Faul, F., & Erdfelder, E. (1992). GPOWER: A priori, post-hoc, and compromise power analyses for MS-DOS [Computer software]. Bonn: Bonn University, Department of Psychology.
Florian, V., & Krulik, T. (1991). Loneliness and social support of mothers of chronically ill children. Social Science and Medicine, 32, 1291-1296.
Frank, R. G., Hagglund, K. J., Schopp, L. H., Thayer, J. F., Vieth, A. Z., Cassidy, J. T., Goldstein, D. E., Beck, N. C., Clay, D. L., Hewett, J. E., Johnson, J. C., Chaney, J. M., & Kashani, J. H. (1998). Disease and family contributors to adaptation in juvenile rheumatoid arthritis and juvenile diabetes. Arthritis Care & Research, 11, 166-176.
Gerhardt, C. A., Vannatta, K., McKellop, J. M., Taylor, J., Passo, M., Reiter-Purtill, J., Zeller, M., & Noll, R. B. (in press). Child-rearing practices of caregivers with and without a child with juvenile rheumatoid arthritis. Journal of Pediatric Psychology.
Harris, J. A., Newcomb, A. F., & Gewanter, H. L. (1991). Psychosocial effects of juvenile rheumatic disease: The family and peer systems as a context for coping. Arthritis Care and Research, 4, 123-130.
Hauser, R. M. (1994). Measuring socioeconomic status in studies of child development. Child Development, 65, 1541-1545.[CrossRef][Web of Science][Medline]
Hochberg, M. C., Chang, R. W., Dwosh, I., Lindsey, S., Pincus, T., & Wolfe, F. (1992). The American College of Rheumatology 1991 revised criteria for the classification of global functional status in rheumatoid arthritis. Arthritis and Rheumatism, 35, 498-502.[Web of Science][Medline]
Holland, B. S., & Copenhaver, M. D. (1988). Improved Bonferroni-type multiple testing procedures. Psychological Bulletin, 104, 145-149.[CrossRef][Web of Science]
Holm, S. (1979). A simple sequentially rejective multiple test procedure. Scandinavian Journal of Statistics, 6, 65-70.
Ireys, H. T., Sills, E. M., Kolodner, K. B., & Walsh, B. B.
(1996). A social support intervention for parents of chidlren
with juvenile rheumatoid arthritis: Results of a randomized trial.
Journal of Pediatric Psychology,
21, 633-641.
Jessop, D. J., Reissman, C. K., & Stein, R. E. (1988). Chronic childhood illness and maternal mental health. Journal of Developmental and Behavioral Pediatrics, 9, 147-156.[Web of Science][Medline]
Kazak, A. E. (1989). Families of chronically ill children: A systems and social-ecological model of adaptation and challenge. Journal of Consulting and Clinical Psychology, 57, 25-30.[CrossRef][Web of Science][Medline]
Kronenberger, W. G., & Thompson, R. J. (1990). Dimensions of family functioning in families with chronically ill children: A higher order factor analysis of the Family Environment Scale. Journal of Clinical Child Psychology, 19, 380-388.[CrossRef][Web of Science]
Kronenberger, W. G., & Thompson, R. J. (1992).
Psychological adaptation of mothers of children with spina bifida: Association
with dimensions of social relationships. Journal of Pediatric
Psychology, 17,
1-14.
Lawrence, R. C., Helmick, C. G., Arnett, F. C., Deyo, R. A., Felson, D. T., Giannini E. H., Heyse, S. P., Hirsch, R., Hochberg, M. C., Hunder, G. G., Liang, M. H., Pillemer, S. R., Steen, V. D., & Wolfe, F. (1998). Estimates of the prevalence of arthritis and selected musculoskeletal disorders in the United States. Athritis & Rheumatism, 41, 778-799.
Loveland-Cherry, C. J., Youngblut, J. M., & Kline Leidy, N. W. (1989). A psychometric analysis of the Family Environment Scale. Nursing Research, 38, 262-266.[Web of Science][Medline]
Lustig, J. L., Ireys, H. T., Sills, E. M., & Walsh, B. B.
(1996). Mental health of mothers of children with juvenile
rheumatoid arthritis: Appraisal as a mediator. Journal of Pediatric
Psychology, 21,
719-733.
McCormick, M. C., Stemmler, M. M., & Athreya, B. (1986). The impact of childhood rheumatic diseases on the family. Arthritis and Rheumatism, 29, 872-879.[Web of Science][Medline]
Masten, A. S. (2001). Ordinary magic: Resilience processes in development. American Psychologist, 56, 227-238.[CrossRef][Medline]
Minden, K., Kiessling, U., Listing, J., Niewerth, M., Doring, E., Meincke, J., Schontube, M., & Zink, A. (2000). Prognosis of patients with juvenile chronic arthritis and juvenile spondyloarthropathy. Journal of Rheumatology, 27, 2256-2263.
Moore, I. M., Gilliss, C. L., & Martinson, I. (1988). Psychosomatic symptoms in parents 2 years after the death of a child with cancer. Nursing Research, 37, 104-107.[Web of Science][Medline]
Moos, R. H., & Moos, B. S. (1994). Family Environment Scale manual: Development, applications, research (3rd ed.). Palo Alto, CA: Consulting Psychologists Press.
Myones, B. L., Williams, G. F., Billings, A., & Miller, J. J. (1988). Social environment in families of children with juvenile arthritis. Arthritis Care and Research, 1, 17-22.
Nakao, K., & Treas, J. (1992). The 1989 Socioeconomic Index of Occupations: Construction from the 1989 Occupational Prestige Scores (General Social Survey Methodological Report No. 74). Chicago: University of Chicago, National Opinion Research Center.
Noll, R. B., Gartstein, M. A., Hawkins, A., Vannatta, K., Davies, W. H., & Bukowski, W. M., (1995). Comparing parental distress for families with children who have cancer and matched comparison families without children with cancer. Family Systems Medicine, 13, 11-28.
Noll, R. B., Kozlowski, K., Gerhardt, C., Vannatta, K., Taylor, J., & Passo, M. (2000). Social, emotional, and behavioral functioning of children with juvenile rheumatoid arthritis. Arthritis & Rheumatism, 43, 1387-1396.[CrossRef][Web of Science][Medline]
Noll, R. B., Ris, M. D., Davies, W. H., Bukowski, W. M., & Koontz, K. (1992). Social interactions between children with cancer or sickle cell disease and their peers: Teacher ratings. Journal of Developmental & Behavioral Pediatrics, 13, 187-193.[Web of Science][Medline]
Noll, R. B., Swiecki, E., Gartstein, M., Vannatta, K., Kalinyak, K. A., Davies, W. H., & Bukowski, W. M. (1994). Parental distress, family conflict, and the role of social support for caregivers with and without a child with sickle cell disease. Family Systems Medicine, 12, 281-294.
Norbeck, J. S., & Anderson, N. J. (1989). Psychosocial predictions of pregnancy outcomes in low-income Black, Hispanic, and White women. Nursing Research, 38, 204-209.[Web of Science][Medline]
Norbeck, J. S., Lindsey, A. M., & Carrieri, V. L. (1981). The development of an instrument to measure social support. Nursing Research, 30, 264-269.[Web of Science][Medline]
Norbeck, J. S., Lindsey, A. M., & Carrieri, V. L. (1983). Further development of the Norbeck Social Support Questionnaire: Normative data and validity testing. Nursing Research, 32, 4-9.[Web of Science][Medline]
Peterson, L. S., Mason, T., Nelson, A. M., O'Fallon, W. M., & Gabriel, S. E. (1997). Psychosocial outcomes and health status of adults who have had juvenile rheumatoid arthritis: A controlled, population-based study. Arthritis & Rheumatism, 40, 2235-2240.[Web of Science][Medline]
Pinals, R. S., Masi, A. T., & Larsen, R. A. (1981). Preliminary criteria for clinical remission in rheumatoid arthritis. Arthritis & Rheumatism, 24, 1308-1315.[Web of Science][Medline]
Phares, V., & Compas, B. E. (1992). The role of fathers in child and adolescent psychopathology: Make room for daddy. Psychological Bulletin, 111, 387-412.[CrossRef][Web of Science][Medline]
Quittner, A. L., Espelage, D. L., Opipari, L. C., Carter, B., Eid, N., & Eigen, H. (1998). Role strain in couples with and without a child with a chronic illness: Associations with marital satisfaction, intimacy, and daily mood. Health Psychology, 17, 112-124.[CrossRef][Web of Science][Medline]
Quittner, A. L., Opipari, L. C., Regoli, M. J., Jacobsen, J., & Eigen, H. (1992). The impact of caregiving and role strain on family life: Comparisons between mothers of children with cystic fibrosis and matched controls. Rehabilitation Psychology, 37, 275-290.[CrossRef]
Reisine, S. T. (1995). Arthritis and the family. Arthritis Care and Research, 8, 265-271.
Roosa, M. W., & Beals, J. (1990). Measurment issues in family assessment: The case of the Family Environment Scale. Family Process, 29, 191-198.[CrossRef][Web of Science][Medline]
Sandberg, D. E., Meyer-Bahlburg, H. F. L., & Yager, T. J. (1991). The Child Behavior Checklist nonclinical standardization samples: Should they be used as norms? Journal of the American Academy of Child and Adolescent Psychiatry, 30, 124-134.[Web of Science][Medline]
Silver, E., Bauman, L., & Ireys, H. (1995). Relationships of self-esteem and efficacy to psychological distress in mothers of children with chronic physical illness. Health Psychology, 14, 333-340.[CrossRef][Web of Science][Medline]
Timko, C., Stovel, K. W., & Moos, R. H. (1992). Functioning among mothers and fathers of children with juvenile rheumatic disease: A longitudinal study. Journal of Pediatric Psychology, 6, 705-724.
Vandvik, I. H., & Eckblad, G. (1991). Mothers of children with recent onset of rheumatic disease: Associations between maternal distress, psychosocial variables, and the disease of the children. Developmental and Behavioral Pediatrics, 12, 84-91.
Vandvik, I. H., Hoyeraal, H. M., & Fagertun, H. (1989). Chronic family difficulties and stressful life events in recent onset juvenile arthritis. Journal of Rheumatology, 16, 1088-1092.
Wallander, J. L., & Varni, J. W. (1998). Effects of pediatric chronic physical disorders on child and family adjustment. Journal of Child Psychology and Psychiatry, 39, 29-46.[CrossRef][Web of Science][Medline]
Zak, M., & Pedersen, F. K. (2000). Juvenile
chronic arthritis into adulthood: A long-term follow-up study.
Rheumatology, 39,
198-204.
CiteULike 
Connotea 
Del.icio.us What's this?
This article has been cited by other articles:
|
|
 |
|
  J. Reiter-Purtill, E. K. Schorry, A. M. Lovell, K. Vannatta, C. A. Gerhardt, and R. B. Noll Parental Distress, Family Functioning, and Social Support in Families with and without a Child with Neurofibromatosis 1 J. Pediatr. Psychol., May 1, 2008; 33(4): 422 - 434. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
R. T. Brown, L. Wiener, M. J. Kupst, T. Brennan, R. Behrman, B. E. Compas, T. David Elkin, D. L. Fairclough, S. Friebert, E. Katz, et al. Single Parents of Children with Chronic Illness: An Understudied Phenomenon J. Pediatr. Psychol., May 1, 2008; 33(4): 408 - 421. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
N. R. Andrews, J. M. Chaney, L. L. Mullins, J. L. Wagner, K. A. Hommel, and J. N. Jarvis Brief Report: Illness Intrusiveness and Adjustment among Native American and Caucasian Parents of Children with Juvenile Rheumatic Diseases J. Pediatr. Psychol., November 1, 2007; 32(10): 1259 - 1263. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
M. J. Bonner, K. K. Hardy, V. W. Willard, and K. C. Hutchinson Brief Report: Psychosocial Functioning of Fathers as Primary Caregivers of Pediatric Oncology Patients J. Pediatr. Psychol., August 1, 2007; 32(7): 851 - 856. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
K. E. Robinson, C. A. Gerhardt, K. Vannatta, and R. B. Noll Parent and Family Factors Associated with Child Adjustment to Pediatric Cancer J. Pediatr. Psychol., May 1, 2007; 32(4): 400 - 410. [Abstract] [Full Text] [PDF]
|
|
|
|
 |
|
 R. Banthia, J. T. Moskowitz, M. Acree, and S. Folkman Socioeconomic Differences in the Effects of Prayer on Physical Symptoms and Quality of Life J Health Psychol, March 1, 2007; 12(2): 249 - 260. [Abstract] [PDF]
|
|
|
|
|
|
R. Riddle, C. N. Ryser, A. A. Morton, J. D. Sampson, R. H. Browne, M. G. Punaro, and R. J. Gatchel The Impact on Health-Related Quality of Life from Non-Steroidal Anti-Inflammatory Drugs, Methotrexate, or Steroids in Treatment for Juvenile Idiopathic Arthritis J. Pediatr. Psychol., April 1, 2006; 31(3): 262 - 271. [Abstract] [Full Text] [PDF]
|
|
|
|
|
|
C. A. Gerhardt, K. Vannatta, J. M. McKellop, J. Taylor, M. Passo, J. Reiter-Purtill, M. Zeller, and R. B. Noll Brief Report: Child-Rearing Practices of Caregivers With and Without a Child With Juvenile Rheumatoid Arthritis: Perspectives of Caregivers and Professionals J. Pediatr. Psychol., June 1, 2003; 28(4): 275 - 279. [Abstract] [Full Text] [PDF]
|
|
| ||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||||