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CLINICAL INVESTIGATION

The Health Education for Lupus Study: A Randomized


Controlled Cognitive-Behavioral Intervention Targeting
Psychosocial Adjustment and Quality of Life in Adolescent
Females With Systemic Lupus Erythematosus
Ronald T. Brown, PhD, Stephanie R. Shaftman, MSc, MS, Barbara C. Tilley, PhD, Kelly K. Anthony, PhD,
Mary C. Kral, PhD, Bonnie Maxson, MA, Laura Mee, PhD, Melanie J. Bonner, PhD, Larry B. Vogler, MD,
Laura E. Schanberg, MD, Mark A. Connelly, PhD, Janelle L. Wagner, PhD, Richard M. Silver, MD
and Paul J. Nietert, PhD

Abstract: Introduction: To examine in a randomize controlled onset SLE represents 15% to 20% of all cases, with a median age
feasibility clinical trial the efficacy of a cognitive-behavioral interven- of onset between 10 and 12 years. Females are affected 3 to 5
tion designed to manage pain, enhance disease adjustment and times more than males, and African American females are dis-
adaptation and improve quality of life among female adolescents with proportionately affected, when compared with American whites.2
systemic lupus erythematosus. Methods: Female adolescents (n 5 53) Clinical manifestations of SLE are extremely variable and unpre-
ranging in age from 12 to 18 years were randomly assigned to 1 of 3 dictable and may include neuropsychiatric symptoms (anxiety
groups including a cognitive-behavioral intervention, an education-only and depression, cognitive deficits and psychosis), fatigue, skin
arm and a no-contact control group. Participants were assessed at base- rashes, arthralgia, headache, seizures, cerebrovascular accidents,
line, postintervention and at 3- and 6-month intervals after completion movement disorders and neuropathy. The musculoskeletal
of the intervention. Results: No significant differences were revealed system is frequently affected, resulting in arthritis that may be
among the 3 treatment arms for any of the dependent measures at any extremely painful. In fact, a high frequency of pain episodes in
of the assessment points. For the mediator variables, a posthoc adolescents with SLE independently has been associated with
secondary analysis did reveal increases in coping skills from baseline fatigue. In addition, the presence of pain among these adoles-
to postintervention among the participants in the cognitive-behavioral cents has been inversely related to quality of life.3 The unpredict-
intervention group compared with both the no-contact control group and able disease course of SLE including disease exacerbations or
the education-only group. Conclusion: Although no differences were flares and episodes of pain that may occur irrespective of health-
detected in the primary outcome, a possible effect on coping of female related behaviors may provoke feelings of learned helplessness
adolescents with systemic lupus erythematosus was detected in this and anxiety. The disease course in juvenile-onset SLE is typi-
feasibility study. Whether the impact of training in the area of coping cally more severe, when compared with adult-onset SLE, char-
was of sufficient magnitude to generalize to other areas of functioning, acterized by more aggressive renal and hematologic disease and
such as adjustment and adaptation, is unclear. Future phase III higher requirement for steroids and immunosuppressive drugs.4–6
randomized trials will be needed to assess additional coping models Juvenile-onset SLE most commonly presents during
and to evaluate the dose of training and its influence on pain adolescence, a time of rapid physical, emotional and social
management, adjustment and health-related quality of life. changes. For adolescents diagnosed with SLE, coping with a
chronic illness may compromise attainment of developmentally
Key Indexing Terms: Lupus; Cognitive-behavioral; Quality of life.
appropriate “tasks” that include increased autonomy, greater
[Am J Med Sci 2012;344(4):274–282.]
investment in social relationships and concern about peer accep-
tance and perceived mastery.7 The unpredictable course of SLE,
S ystemic lupus erythematosus (SLE) is an episodic, multi-
system, autoimmune disease characterized by widespread
inflammation of blood vessels and connective tissue.1 Juvenile-
including disease exacerbations that may occur irrespective of
health-related behaviors, may provoke feelings of learned help-
lessness and anxiety. Managing medical regimens and physician
visits may perpetuate dependency on adults at a time when
From the Department of Psychology (RTB), Wayne State University, increased autonomy is desired. Symptoms that affect appearance
Detroit, Michigan; Department of Epidemiology and Biometry (SRS), Med- (eg, skin rashes, facial swelling, hair loss, weight gain and easy
ical University of South Carolina, Charleston, South Carolina; Department
of Epidemiology and Biometry (BCT), University of Texas Health Sciences bruising) may fuel feelings of self-consciousness and social
Center, Houston, Texas; Department of Psychiatry (KKA, MJB), Duke Univer- isolation. Not surprisingly, many adolescents with SLE struggle
sity Medical Center, Durham, North Carolina; Department of Pediatrics with depression.8 Many youth with rhetmatologic disorders,
(MCK), Medical University of South Carolina, Charleston, South Carolina; including SLE, also report greater concern about peer relation-
Department of Psychiatry (BM, LM), Emory University School of Medicine,
Atlanta, Georgia; Department of Pediatrics (LBV), Emory University School ships and self-concept, when compared with health-related
of Medicine, Atlanta, Georgia; Department of Pediatrics (LES), Duke Uni- matters, such as activities of daily living and general physical
versity Medical Center, Durham, North Carolina; Department of Pediatrics health.9 As such, maladaptive psychosocial responses to chronic
(MAC), University of Missouri Kansas City, Kansas City, Missouri; and
Department of Medicine (JLW, RMS, PJN), Medical University of South
illness may in turn adversely impact healthcare utilization and
Carolina, Charleston, South Carolina. adherence to complicated medical regimens.
Submitted June 18, 2011; accepted in revised form October 26, 2011. Finally, another line of research has demonstrated that
This study was supported by a grant award from the National Institute biologic processes (eg, severity of disease and functional
for Arthritis and Musculoskeletal and Skin Diseases, 1 P60 AR049459-01.
Correspondence: Ronald T. Brown, PhD, Office of the Provost, Wayne
outcome) mediate the health outcomes of various diseases,
State University, 656 West Kirby, Detroit, MI 48202 (E-mail: rtbrown@ including quality of life.10 Specifically, interventions that
wayne.edu). enhance disease adaptation and adjustment also may be affected


274 The American Journal of the Medical Sciences Volume 344, Number 4, October 2012
Health Education for Patients With Lupus

by specific confounders or effect mediators, including disease important to note that this investigation used a novel delivery
severity and functional outcomes. For example, the effect of system by home-based completion of computerized modules,
remediating coping strategies characterized by negative think- thereby examining the feasibility of a service delivery model
ing, catastrophizing and self-statements of fear in children has that could potentially reach many underserved families.
not been studied. The efficacy of an intervention is important in
understanding whether negative cognitions may be modified METHODS
and whether disease adjustment and disease adaptation may This study began in 2003 as a multicenter, phase III trial
be enhanced. Finally, there is extensive evidence in the litera- with an expected enrollment of 147 participants and 3 treatment
ture concerning the buffering effects of peer and family social arms—a CBT group, an education-only group and a no-contact
support on adolescents’ disease adaptation and adjustment.10 control group—using a 1:1:1 randomization scheme. As time
The role of social support as a mediator of an intervention progressed, participant recruitment became problematic, and the
program on adolescents’ adjustment and health quality of life investigation was subsequently redesigned as a phase II feasi-
remains unclear.10 bility study. The randomization allocation was changed to 2:0:1
Clearly, the chronic, debilitating course of SLE presents (CBT, education-only and no-contact control), and it was
tremendous challenges to the individual’s coping resources. decided that the primary comparison would be between the
However, psychosocial adjustment to SLE has been understudied CBT group and the no-contact control group at the 7-week
and poorly understood,11,12 especially in juvenile-onset SLE. In assessment. Comparisons to the education-only patients already
a randomized controlled clinical trial, Navarrete-Mavarrete et al13 recruited to the study (n 5 10) would be considered secondary.
examined the efficacy of a cognitive-behavioral intervention in Figure 1 illustrates the flow of patients throughout the study.
a group of adult patients with lupus. Participants were 45 adult
patients with lupus who were characterized by high levels of Participants
distress. Participants were assigned randomly to a control group Participants were recruited from 3 major southeastern
or an active therapy group consisting of cognitive-behavioral medical centers (Duke University, Emory University and
therapy (CBT). Dependent measures included assessments of Medical University of South Carolina) after the institutional
psychologic functioning, disease severity and quality of life. review boards at each institution approved the study. Because of
Findings revealed that symptoms of depression and anxiety and the preponderance of SLE among females, only adolescent
daily stress decreased for the patients in the active cognitive females were included in the study. Participants ranging in age
therapy group relative to the control group; increased quality of from 12 to 18 years who had an existing diagnosis of SLE as
life also was reported for those participating in the active ther- defined by American College of Rheumatology criteria15 were
apy condition. On the basis of their findings, it is concluded included in the study. The majority of the adolescents in the
that cognitive therapy is an effective intervention in managing sample were African American. An adolescent was excluded
psychologic distress for adult patients suffering from lupus. if she attained a score less than 24 on the Mini Mental Status
Although the data from this investigation are especially encour- Examination16; had severe intellectual impairment as reported by
aging with respect to cognitive therapy as a viable psychologic their caregivers (children with mild intellectual impairments or
treatment for adult patients with SLE, no studies have yet learning disabilities and/or those receiving special education
examined the viability of cognitive therapy for adolescents services as reported by caregivers were still eligible for partici-
with lupus. To date, there have been no published trials of pation in the investigation); had a terminal illness with a life
behavioral interventions specifically designed for adolescents expectancy of less than 1 year; had a lack of facility with the
with SLE. English language or was already enrolled in another clinical trial.
This investigation represents the first known home-based
delivery randomized controlled trial examining the effective- Measures
ness of CBT targeting pain management, enhanced psychoso- The assessment battery in this study was chosen to assess
cial adjustment and improved quality of life for adolescent participants’ functioning with relation to pain severity, adjust-
females with SLE. In recent years, there has been a burgeoning ment and adaptation and health-related quality of life. Each
of interactive and communication technologies for the purpose psychometrically sound-dependent measure was administered
of delivery of psychologic treatments to children and adoles- at each of the 4 data collection visits. A number of assessment
cents.14 Intervention programs that are delivered by means of instruments also were used to assess potential confounders or
interactive technology have the advantage of offering youth effect mediators in the final analyses, including demographic
treatments that are novel, portable and cost effective. Although variables, baseline clinical characteristics and baseline assess-
much research has been conducted on electronic technology ment of social support and coping strategies. Table 1 summa-
with adult populations, the pediatric literature has only recently rizes demographic data for participants in each of the study
emerged. Randomized controlled trials to evaluate internet groups at baseline.
interventions for pediatric populations have shown particular
promise for various health-related conditions. In particular, Dependent Measures
CBTs delivered by the internet also have demonstrated consid- The McGill Pain Questionnaire-Short Form17 was
erable promise for various health conditions.14 Thus, another administered to assess intensity of pain. This study used both
purpose of this investigation was to examine the efficacy of the sensory and the affective dimensions of pain and present
a cognitive-behavioral intervention that was administered by pain intensity.
means of computer-based technology to adolescents with lupus The Behavior Assessment System for Children
and their caregivers. (BASC)18 was chosen as a measure of adjustment and adapta-
It was hypothesized that participants randomly assigned tion because of its excellent psychometric characteristics19 and
to the CBT arm would demonstrate enhanced adjustment (ie, the provision of comparable parent and adolescent forms
improved coping skills, self-efficacy and pain management), (BASC-SRP). The BASC measures multiple aspects of adoles-
when compared with participants randomly assigned to the cent functioning and behavior, including both positive (adap-
education only arm or the wait list control arm. It is also tive) and pathologic (clinical) dimensions. The Internalizing,

Ó 2012 Lippincott Williams & Wilkins 275


Brown et al

FIGURE 1. Flow of participants through the various phases of the study.

Externalizing and Adaptive Behavior Index scores were used in scale that assesses signs and symptoms present in the past
the analyses. 10 days. Scores can range from 0 (no activity) to 105. Partic-
The Positive and Negative Affect Schedule-Extended ipants’ rheumatologists completed the SLEDAI at each of the
Version20 is designed to measure higher and lower order factors 4 data collection visits. The SLEDAI has demonstrated high
associated with the 2-factor structure of affect, positive affec- sensitivity to clinical change in patients with juvenile-onset
tivity and negative affectivity. These factors were used in the SLE.25
analyses. Both the Perceived Social Support (PSS)-Family and
The Self-Perception Profile for Adolescents21 taps global PSS-Friend26,27 were used to assess adolescents’ perceived
self-worth and provides a measure of perceived self-competence. social support from family and friends. These instruments were
For this study, the total perceived self-confidence score was used chosen for this study as they assess functional self-support
in the analyses. practices (eg, coping and remaining functional despite symp-
The Multidimensional Health Locus of Control Scale22 toms). The PSS-Family and PSS-Friend were administered to
assesses attribute of health outcomes to internal forces (ie, one- the adolescents before study enrollment and at the baseline
self), powerful others (eg, family and healthcare providers) or assessment.
chance occurrences (ie, luck). The internal locus of control sub- The Coping Strategies Questionnaire (CSQ)28 was used
scale was used in this study as a means of operationally defining in this study to assess adolescents’ coping primarily as it is
self-efficacy. related to pain. We have chosen pain as the exemplar for coping
The Pediatric Health-Related Quality of Life, Pediatric because it is a tangible manifestation of SLE and other rheuma-
Self-Report and Parent Report (PedsQL)23 was chosen to assess tologic disorders (eg, juvenile idiopathic arthritis). The CSQ
health-related quality of life in the core dimensions of physical assesses cognitive, behavioral and physiologic strategies specif-
functioning, emotional functioning, social functioning and ically related to pain.29,30 The CSQ was administered to the
school functioning. The pediatric self-report form and the par- adolescents before study enrollment and at the baseline
ent proxy report of child PedsQL Rheumatology Health-Related assessment.
Quality of Life23 also were used for the purpose of complement-
ing the generic core scale. Procedures
After having attained consent from caregivers and assent
Mediator Variables from the adolescents, participants were randomly assigned to
Data also were collected to assess potential confounders 1 of 3 treatment arms.
or effect mediators, including specific demographic variables
(adolescents’ chronological age, health insurance and parent/ CBT Condition
caregiver education level), specific disease characteristics, num- The specific ingredients of the CBT package included
ber of clinic/emergency room/inpatient contacts within the 3 coping skills training and cognitive restructuring techniques.31
months before study enrollment and baseline assessment of For this treatment arm, coping skills training included enhanc-
social support and coping strategies. ing existing skills and teaching new skills to manage stressors
The Systemic Lupus Erythematosus Disease Activity associated with SLE. Efforts to teach active coping skills
Index (SLEDAI)24 was used as a means of assessing disease included training in relaxation, distraction and problem-solving
activity/severity. The SLEDAI is a physician-completed rating skills. The CBT arm also included cognitive restructuring

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TABLE 1. Participant characteristics for each of the 3 groups at baseline


Cognitive-behavioral Education-only No-contact control
intervention (n 5 27) (n 5 10) (n 5 16)
Race (% African American) 74 90 81
Age, mean (SD) 15.4 (3.0) 15.0 (3.3)a 15.9 (2.0)
Parent’s education
#High school 33 30 56
Some college 37 20 19
College graduate 30 50 25
Health insurance coverage (% yes) 93a 100a 75
Years since diagnosis of SLE, mean (SD) 3.0 (2.6) 3.3 (2.2)a 2.0 (1.9)
Baseline SLEDAI disease activity score
Mean (SD) 4.7 (4.2) 3.4 (2.8) 6.1 (9.6)
$6 (%) 37 20 38
In 3 months before baseline visit
Inpatient hospital visit (% yes) 8a 0a 38
Emergency department visit (% yes) 8a 10a 38
Outpatient clinic visits, median 1.5 (0–3) 1.5 (1–4) 1.5 (1–3.5)
SLE flare events, median 0 (0–1) 0 (0–0) 0 (0–1)
SLE flare event (% yes) 35 20 38
Perceived Social Support Scale
Family score 15 (9–19) 17 (14–19) 14 (11–18)
Friend score 15 (10–18) 16 (14–18) 14 (13–18)
Coping Strategies Questionnaire
Catastrophizing, median 14 (10–20)a 12 (5–17) 9 (8–29)
Coping self-statements, median 20 (14–26)a 26 (19–36) 26 (21–33)
Diverting attention, median 17 (14–20) 19 (8–34) 20 (12–23)
Increase behavioral activities, median 16 (12–22) 23 (11–34) 17 (12–24)
Ignoring sensations, median 13 (8–18) 17 (9–36) 18 (4–25)
Praying/hoping, median 28 (20–30) 27 (19–35) 29 (26–33)
Reinterpreting pain sensations, median 10 (4–17) 10 (3–24) 12 (2–18)
a
P , 0.15 compared with no-contact control group by x2 test, Fisher’s exact test or Wilcoxon’s rank sum test and thus included as a covariate in
the relevant multivariable analyses.
SLE, systemic lupus erythematosus; SLEDAI, Systemic Lupus Erythematosus Disease Activity Index; SD, standard deviation.

exercises designed to focus on negative cognitions and evalu- after the third session to monitor treatment adherence, answer
ative responses to stressful situations in daily life, negative any questions about the module and ensure compliance with
perceptions of physical appearance and negative responses to treatment homework. Each lesson took approximately 45 minutes
pain. to complete. Primary caregivers also were provided with written
The CBT arm was administered during the course of materials about the intervention and tips regarding ways to
6 weeks. Because of the extant psychosocial intervention support and encourage their adolescent in the acquisition and
literature underscoring the importance of time-limited and practice of the skills taught during each module.
focused interventions that may be delivered during a period The first session focused on established rapport with the
of several weeks and because of the numerous medical adolescent, explaining the rationale for the cognitive-behavioral
appointments and ongoing follow-up for these adolescents, intervention and teaching self-monitoring of mood and activity
we opted for a treatment period that was relatively short term. level. The second session focused on coping skills. This
Short-term therapies have been fairly typical in the cognitive included training in relaxation (eg, progressive muscle relaxa-
therapy literature with pediatric populations.32 In addition, tion and diaphragmatic breathing) and distraction (eg, focusing
shorter interventions are typically associated with fewer com- on 1 thing in the physical environment and mental counting
pliance problems.10 techniques). In addition, an important component addressed
The participants were provided with laptop computers to problem-solving skills (eg, appropriate pacing of daily activi-
take home for use during the intervention. The computer had ties). These relaxation, distraction and problem-solving skills
3 separate manualized modules preinstalled that were password were discussed during the session, and the adolescents were
protected. In weeks 1, 3 and 5, the participants worked on asked to practice and apply skills as homework between
a separate module containing several lessons. Participants were sessions. Obstacles to practicing skills at home were discussed
required to complete each of the lessons during the week after as a means of relapse prevention.
completion of a module. A member of the research team Finally, the third session focused on calming self-
telephoned the participants in the week between sessions statements and cognitive restructuring that were designed to
1 and 2, sessions 2 and 3 and during the week immediately diminish negative affectivity and alter perceptions of pain.

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Brown et al

Cognitive restructuring also was used for the purpose of altering For missing data, we used a worst-case-scenario approach,33
distorted perceptions of physical appearance and social which involved assigning values for outcomes equal to the
competence. Again, strategies were described, presented on worst outcome among participants in their treatment group at
a CD-ROM and practiced with the adolescents. In this session, the time point of interest.
additional training was provided in the area of relapse pre- Because there were multiple correlated outcome meas-
vention. Adolescents were required to complete a record of their ures, we used a global statistical test (GST)34 statistic as the
practice. Self-monitoring was used as a means of ensuring dependent variable in an analysis of covariance (ANCOVA)
compliance. The therapists also conducted 2 10- to 15-minute model to compare outcomes between treatment groups. To
telephone contacts in the week between sessions and in the compute the GST, each participant’s relative rankings on
week immediately after the third session to review their individual outcomes (domains) were computed, summed and
homework assignment and assure compliance with treatment. averaged by the number of outcomes, which gave each partic-
For any participant who experienced an acute flare of lupus ipant a single summary score. For each treatment group, the
symptoms, the intervention sessions were postponed until the mean rank differences, associated 95% confidence intervals and
adolescent was designated stable by the site rheumatologist. P values comparing the treatment groups were reported. Aside
Booster sessions (sessions 4 and 5) were held with each from not making any assumptions about the underlying distri-
adolescent 2 and 5 months after they began the study. These bution of the outcome measures, a benefit of using ranks for
sessions focused on continued cognitive skill development, reporting across such a varied list is the inherent standardization
application, refinement and relapse prevention beginning with that occurs. The domains that were included in the GST were
a discussion of the patient’s current status and use of coping (1) personal adjustment index (BASC-SRP-adolescent form),
skills. Although a complete description of the intervention is (2) affectivity (Positive and Negative Affect Schedule-Extended
not possible within the space constraints of this article, the Version), (3) perceptions of physical appearance and social
complete instructional manual and modules are available from competence (Self-Perception Profile for Adolescents), (4) pain
the senior author by request. intensity (McGill Pain Questionnaire-Short Form), (5) quality
of life (Peds-QL and Peds-QL Rheumatology) and (6) self-
Education-Only Condition efficacy (Multidimensional Health Locus of Control Scale).
An education-only intervention was administered in The GST represents sums of the participants’ rankings of their
a similar format to the intervention arm outlined earlier. changes from baseline to follow-up assessment for each of the
Participants in this condition who were recruited from each of various outcomes.
the 3 sites were provided with the rationale that understanding In the ANCOVA model, the treatment arm served as the
their disease would help them manage the disease more independent variable of interest, with covariate adjustment as
appropriately. As in the active-intervention arm, sessions in needed. Separate ANCOVA models were created to perform
both the active and maintenance phases of this intervention primary and secondary comparisons for each domain included in
were presented as 45-minute long modules on a laptop the GST. The 3- and 6-month outcomes were included in the
computer. The first session focused on the impact of SLE on secondary analyses to determine the durability/sustainability of
daily functioning at home and at school. The second session, treatment. Our study was designed to provide 90% power for
held during the third week, focused on issues pertaining to detecting a moderate effect size (Cohen’s d 5 0.64) for the
healthy living (eg, maintaining a balanced diet, exercise and primary comparison between the CBT group and no-contact
appropriate sleep hygiene). The third session, conducted during control group, assuming an alpha of 0.25 and 1-sided hypothesis
the fifth week, reviewed the material that had been taught up testing, a strategy used for pilot studies of this type.35 Although
to that point. In addition, the therapist also conducted 2 10- to the education-only group was originally part of the design as an
15-minute telephone contacts during the week between the additional control, recruitment difficulties required us to focus on
first and second sessions, the second and third sessions and the other 2 groups. Thus, the power to detect a moderate effect
the week immediately after the third session to review the size between the education-only group and the other groups was
instructional materials. somewhat lower (intervention group: 85% and no-contact con-
trol group: 82%), with a 5 0.25 and 1-sided hypothesis testing.
No-Contact Control Condition Finally, posthoc analyses (Wilcoxon’s rank sum tests)
A no-contact control group recruited equally from the were performed to test whether there were any significant differ-
3 sites was included in this study for the purpose of eliminating ences by treatment group in the changes in the subscores of the
possible secular effects over time on the dependent variables. CSQ. The CSQ subscores served as process measures, and their
Any differences in participants’ characteristics at baseline (ie, analyses helped to determine whether participants were, in fact,
years since SLE diagnosis, inpatient hospital visits, emergency altering their coping strategies in response to the intervention.
department visits and insurance coverage) were covaried in the
final analysis.
For each of the 3 treatment conditions, the assessment RESULTS
battery was administered immediately before the intervention Participants’ demographic and baseline characteristics
(baseline), immediately after the intervention (ie, post-test), are summarized in Table 1. Each of the groups was similar with
7 weeks after the commencement of the intervention and finally respect to race, age, parents’ education level, years since
at 3 and 6 months after the intervention program. For each study diagnosis of SLE, baseline disease activity, recent healthcare
visit, participants were offered a nominal monetary incentive to services, social support and most coping strategies. Compared
defray the cost of gasoline or other travel expense. with participants in the no-contact control condition, partici-
pants in the active intervention arm were more likely to have
Statistical Analyses health-insurance coverage (93% versus 75%, P , 0.15), fewer
Because some attrition was expected, the primary data visits to the emergency department during the 3 months before
analyses were conducted using an intention-to-treat approach, randomization (8% versus 38%, P , 0.15) and higher (P ,
including all participants who were randomized in the analyses. 0.15) catastrophizing and lower coping self-statement scores.

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Finally, compared with the participants in the no-contact control post-testing revealed that the CBT group did not exhibit
group, participants in the education-only arm were younger significant overall improvement compared with the no-contact
(15.0 years versus 15.9 years, P , 0.15), more likely to have control group (T 5 –0.34, P 5 0.63) (Table 3). Secondary
health insurance (100% versus 75%, P , 0.15), diagnosed with analyses revealed that the performance of the education-only
lupus for a longer period of time (3.3 versus 2.0 years, P , 0.15) arm was somewhat better than the no-contact control group at
and had fewer visits to the emergency department during the the 3-month follow-up (T 5 1.24, P 5 0.11) and at the 6-month
3 months before randomization (10% versus 38%, P , 0.15). follow-up (T 5 0.92, P 5 0.19), where the levels of statistical
A summary of participant and caregiver report at significance fell below the 0.25 threshold, which had been the
baseline on the outcome measures for each of the 3 groups is alpha level prespecified for the primary analyses.
presented in Table 2. The analyses of changes from baseline to post-testing on
Results of the primary analysis of the GST that the CSQ suggest that the patterns of change in coping behaviors
compared the CBT group and the no-contact control group at were significantly different among the 3 treatment arms (Table 4).

TABLE 2. Baseline data for the primary outcome measures


Cognitive-behavioral Education-only No-contact control
intervention (n 5 27) (n 5 10) (n 5 16)
Measures M SD M SD M SD
SF-MPQ
Sensory dimension 4.84 5.50 5.50 6.19 5.88 8.52
Affective dimension 2.30 2.37 1.90 2.77 1.25 1.34
Present pain intensity (PPI) 9.52 9.25 8.60 9.19 9.94 12.01
BASC-PRS
Internalizing Problems Index 57.81 8.67 52.00 8.19 58.25 14.13
Externalizing Problems Index 48.07 11.32 45.80 5.45 44.75 10.34
Adaptive Behavior Index 47.63 10.18 53.30 7.75 51.69 11.33
BASC-SRP
Emotional Symptoms Index 49.33 10.39 45.60 8.53 46.94 7.17
Clinical Maladjustment Index 49.37 6.90 47.40 7.29 46.94 7.71
School Maladjustment Index 42.70 7.26 45.10 4.51 46.38 7.86
Personal Adjustment Index 50.56 12.42 52.10 10.08 50.81 7.34
PANAS-X
Positive affectivity 31.78 9.97 33.30 11.93 31.63 8.81
Negative affectivity 13.63 3.59 12.20 3.08 12.00 3.06
SPPA
Physical appearance 13.48 4.52 13.50 4.50 15.06 3.66
Self-worth 16.26 3.48 15.20 3.91 17.63 1.82
Social acceptance 14.40 4.72 16.15 3.08 15.81 3.33
MHLC
Internal locus of control, form B 22.31 6.79 27.60 2.84 22.44 6.26
Internal locus of control, form C 16.88 6.51 22.90 2.92 17.98 6.59
PedsQL Teen Self-Report Total Score 62.65 18.54 69.21 16.40 68.53 15.51
Peds QL Parent Report Total Score 61.42 9.87 77.76 10.05 63.74 21.06
Peds QL Rheumatology Teen Self-Report
Daily activities 77.04 23.34 89.00 13.50 81.25 21.17
Communication 58.95 29.14 54.17 25.53 69.27 21.02
Pain and hurt 48.38 26.89 74.38 21.74 53.52 40.70
Treatment 71.30 26.07 82.50 16.53 73.21 15.15
Worry 50.93 31.38 55.83 30.69 57.29 27.20
Peds QL Rheumatology Parent Report
Daily activities 80.10 24.57 90.00 14.42 78.78 28.36
Communication 69.38 28.58 83.33 20.41 69.05 35.87
Pain and hurt 45.35 20.35 70.63 31.74 51.53 28.98
Treatment 72.99 22.36 91.14 18.62 76.81 20.70
Worry 64.50 25.55 84.26 24.45 66.96 33.03
SF-MPQ, McGill Pain Questionnaire-Short Form; BASC, Behavior Assessment System for Children; PRS, Parent Rating Scale; SRP, Self-Report
of Personality; PANAS-X, Positive and Negative Affect Schedule-Extended Version; SPPA, Harter Self-Perception Profile for Adolescents; MHLC,
Multidimensional Health Locus of Control Scales; PedsQL, Pediatric Health-Related Quality of Life, Pediatric Self-Report and Parent Report; Peds
QL Rheumatology, Pediatric Rheumatology Health-Related Quality of Life.

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Brown et al

TABLE 3. Summary of primary and secondary analyses


Comparison of Interest t df Pa
Primary analysis
Cognitive-behavioral intervention (n 5 27) vs. no-contact control (n 5 16), post-test –0.34 36 0.63
Secondary analyses
Cognitive-behavioral intervention (n 5 27) vs. no-contact control (n 5 16), 3-month follow-up –0.39 36 0.65
Cognitive-behavioral intervention (n 5 27) vs. no-contact control (n 5 16), 6-month follow-up –0.74 36 0.77
Education-only (n 5 10) vs. no-contact control (n 5 16), post-test –0.80 19 0.78
Education-only (n 5 10) vs. no-contact control (n 5 16), 3-month follow-up 1.24 19 0.11
Education-only (n 5 10) vs. no-contact control (n 5 16), 6-month follow-up 0.92 19 0.19
Cognitive-behavioral intervention and education-only combined (n 5 37) vs. no-contact control –0.48 46 0.68
(n 5 16), post-test
Cognitive-behavioral intervention and education-only combined (n 5 37) vs. no-contact control –0.15 46 0.56
(n 5 16), week 12
Cognitive-behavioral intervention and education-only combined (n 5 37) vs. no-contact control –0.38 46 0.65
(n 5 16), 6-month follow-up
a
The P value reported comes from a 1-sided test of the null hypothesis that the intervention is no better than the control, using an analysis of
covariance model with the global statistical test as the dependent variable and treatment-group assignment as the independent variable, adjusting for
baseline covariates that were significantly (P , 0.15) different between comparison groups.

Specifically, increases in the “coping self-statements” and but better self-efficacy and self-worth for the CBT group relative
“increased behavioral activities” subscores were significantly to the no-contact control group. Parents of adolescents in the
(P , 0.05) greater among the participants in the CBT group CBT group also reported higher frequencies of quality of life
compared with the no-contact control group. Increases in the for their adolescents (0.46) relative to the no-contact control
“coping self-statements” and “diverting attention” subscores group. The effect sizes for adolescents’ negative affectivity
were significantly (P , 0.05) greater among participants in the (–0.32) and physical appearance (–0.39) were in the negative
CBT group compared with the education-only group. direction, indicating that the control group evidenced a greater
Finally, effect sizes were computed between the CBT increase than the intervention group. All other effect sizes were
group and the no-contact control group at the post-test (7 weeks) less than one third of a standard deviation.
follow-up assessment. For each of the effect sizes computed for
the dependent variables, only 1 effect size (self-efficacy and DISCUSSION
physician’s influence) exceeded 1.00, and this was a negative The purpose of this study was to examine the efficacy of
effect size, indicating that the control group evidenced more of a cognitive-behavioral intervention designed to manage pain,
an increase than the intervention group. Moreover, only 1 effect enhance adjustment and adaptation and increase quality of life
size (affective dimension of pain) exceeded one half of a standard among adolescents with SLE. This particular study is unique
deviation (0.54), suggesting a trend for greater self-efficacy and because, to date, it is the first known randomized controlled clinical
less affective dimensions of pain for the CBT group relative to trial designed to enhance adjustment among adolescent females
the no-contact control group. A number of other effect sizes with SLE delivered by means of a home-based completion of
exceeded one third of a standard deviation, including parents’ computer modules. Our study used 3 treatment arms where it was
reports of their adolescents’ externalizing behavior problems hypothesized that participants in the CBT arm would evidence
(0.42), self-efficacy (0.37) and self-worth (0.35), which suggests greater improvement in adjustment relative to those in the
worse externalizing behavior problems as reported by the parents education-only and no-contact control arms. We also hypothesized

Table 4. Changes from baseline to week 7 in coping strategies by treatment group over time
Cognitive-Behavioral No-Contact Control Education-Only Control
Intervention (n527): median (n516): median (n510): median
Coping strategy subscore change (IQR) change (IQR) change (IQR)

Catastrophizing 22 (27 to 4) +1 (24 to 4) 26 (213 to 0)


Coping self-statements +0 (22 to 6)a,b 22 (211 to 1) 26 (213 to 1)
Diverting attention +4 (23 to 10)b +2 (27 to 7) 27 (29 to -3)
Increased behavioral activities +4 (23 to 9)a 23 (213 to 5) 26 (27 to 4)
Ignoring sensations +2 (23 to 8) 21 (25 to 5) 22 (211 to 1)
Praying/hoping 0 (23 to 6) +2 (22 to 8) 22 (25 to 10)
Reinterpreting pain sensations +1 (24 to 13) +1 (24 to 4) 22 (29 to 1)
a
P , 0.05 when compared to the No-Contact group by Wilcoxon rank sum test.
b
P , 0.05 when compared to the Education-Only group by Wilcoxon rank sum test.

280 Volume 344, Number 4, October 2012


Health Education for Patients With Lupus

that social class, disease severity, coping style and the adolescents’ clinical trials that typically recruit the highest functioning
perceived peer and family support would mediate the success of individuals with a particular disease, the participants in this
the CBT intervention. study were certainly no exception. Thus, it is possible that the
For the primary analysis, no significant greater overall participants in this study may have been too high functioning in
improvement was detected for the CBT group compared with the various domains evaluated and thereby evidenced little
the no-contact control group. Similarly, the secondary analyses improvement from the intervention.
showed no significantly greater overall improvement for the The aforementioned limitations notwithstanding, it also
CBT group relative to the no-contact control group at the 3- or should be noted that this was one of the few clinical trials that
6-month follow-ups. The performance of the education-only has been conducted primarily by means of computer technol-
arm was somewhat better across measures than the no-contact ogy, thereby requiring less transportation to and from the
control group at the 3- and 6-month follow-ups. Finally, when medical setting. There is a critical need for novel and effective
the CBT group and education-only group were combined, intervention programs that target psychosocial adjustment
they did not exhibit overall improvement compared with the among youth with chronic illness. Moreover, interventions that
no-contact control group at any of the time points. overcome the barriers of underserved areas are in critical
That the primary analyses failed to confirm our initial shortage. The computerized presentation of the intervention
hypothesis is disappointing. A “floor effect” may account for provided in this clinical trial is easily administered in the home
the failure to detect significant overall improvement in outcome setting, thereby making it cost effective. Although participant
variables, as both adolescent participants and their caregivers recruitment was challenging, once enrolled, no participants
reported nonsignificant levels of maladjustment at baseline. It is were lost to follow-up, lending further support for the feasibility
also possible that the small sample size was responsible for the of this service delivery model.
failure to demonstrate an effect (ie, type II error). It is Although this study fell short of demonstrating efficacy
encouraging that effect sizes ranged from large to moderate, across various areas of functioning, we anticipate that future
which suggests that the intervention demonstrated some effi- research efforts in this area will be productive in documenting
cacy, albeit insignificant, in the areas of pain management, the efficacy of such interventions for enhancing the quality of
adaptive behavior and quality of life. Future clinical trials using life of adolescents with SLE. Replication of the present
larger samples may yield more encouraging results. clinical trial with a greater number of participants will be
Despite the absence of significant differences on important in determining the veracity of the present findings,
the dependent measures as a function of group placement, analyses so as to establish the empirical evidence for cognitive therapy
of change from baseline to post-testing in the area of coping among female adolescents with SLE and ultimately
revealed significant differences among the 3 treatment arms. Effect establish it as an empirically based intervention for adoles-
sizes that were computed between the CBT group and the no- cents with SLE.
contact control group at the post-test assessment suggested
generally larger effect sizes for self-efficacy and less affective REFERENCES
dimensions of pain, with means in favor of the CBT group. Data 1. Iverson GL. The need for psychological services for persons with
also revealed increases in coping self-statements, diverting attention systemic lupus erythematosus. Rehabil Psychol 1995;40:39–49.
and increased behavioral activity for the CBT group compared with
the no-contact control group. Thus, the training in relaxation and 2. Kone-Paut I, Piram M, Guillaume S, et al. Lupus in adolescence.
distraction, and problem-solving skills (eg, appropriate pacing of Lupus 2007;16:606–12.
daily activities), may have enhanced coping (as evidenced by the 3. Dahlquist LM, Nagel MS. Chronic and recurrent pain. In: Roberts M,
adolescents’ reports). editor. Handbook of pediatric psychology. New York: Guilford; 2009.
The findings of this particular study must be interpreted p.153–70.
within the context of the study limitations. First, the difficulties 4. Brunner HI, Bishnoi A, Barron AC, et al. Disease outcomes and
associated with participant recruitment resulted in a relatively ovarian function of childhood-onset systemic lupus erythematosus.
small number of participants in each of the treatment arms. The Lupus 2006;15:198–206.
small sample size likely mitigated significant effects that may
have been observed in a larger sample. Clearly, recruitment of 5. Gutierrez-Suarez R, Ruperto N, Gastaldi R, et al. A proposal for
a pediatric version of the Systemic Lupus International Collaborating
children and adolescents with a low incidence disorder in a
Clinics/American College of Rheumatology Damage Index based on the
clinical trial that requires ongoing contact with the investigative
analysis of 1,015 patients with juvenile-onset systemic lupus erythema-
team is a major challenge. Despite the fact that this study was
tosus. Arthritis Rheum 2006;54:2989–96.
a multisite clinical trial, the study did not escape the problem
of enrolling a sufficient number of participants. Any future study 6. Bader-Meunier B, Armengaud JB, Haddad E, et al. Initial presen-
would require a much larger number of sites, so as to obtain tation of childhood-onset systemic lupus erythematosus: a French mul-
a greater number of participants to participate in the clinical trial. ticenter study. J Pediatr 2005;146:648–53.
Another limitation of this study may be associated with 7. Beresford MW, Davidson JE. Adolescent development and SLE. Best
the intensity or dose of the intervention, which may not have Practice Res Clin Rheumatol 2006;20:353–68.
been sufficiently large in length or intensity to produce the
8. Nery FG, Borba EF, Hatch JP, et al. Major depressive disorder and
hypothesized changes in the dependent measures. It is recom- disease activity in systemic lupus erythematosus. Compr Psychiatry
mended that future research efforts focus on comparing inter- 2007;48:14–99.
vention programs that differ in dose or intensity of intervention.
In addition, which aspect of the CBT intervention resulted in 9. Taylor J, Passo MH, Champion VL. School problems and teacher
improved coping cannot be determined from this multipush responsibilities in juvenile rheumatoid arthritis. J Sch Health 1987;57:
intervention. Future studies will need to focus on dismantling 186–90.
the various components of treatment used in this study to 10. La Greca AM, Mackey ER. Adherence to pediatric treatment
determine the specific ingredients that actually produced change regimens. In: Roberts M, editor. Handbook of pediatric psychology.
in the area of coping. Finally, consistent with the majority of New York: Guilford; 2009.

Ó 2012 Lippincott Williams & Wilkins 281


Brown et al

11. Dobkin PL, Fortin PR, Joseph L, et al. Psychosocial contributions to 24. Liang MH, Socher SA, Larson MG, et al. Reliability and validity of
mental and physical health in patients with systemic lupus erythemato- six systems for the clinical assessment of disease activity in systemic
sus. Arthritis Care Res 1998;11:23–31. lupus erythematosus. Arthritis Rheum 1989;32:1107–18.
12. Dobkin PL, Da Costa D, Fortin PR, et al. Living with lupus: a pro- 25. Brunner HI, Feldman BM, Bombardier C, et al. Sensitivity of the
spective pan-Canadian study. J Rheumatol 2001;28:2442–8. systemic lupus erythematosus disease activity index, British Isles lupus
assessment group index, and systemic lupus activity measure in the
13. Navarrete-Mavarrete N, Peralta-Ramirez MI, Sabio-Sanchez JM,
evaluation of clinical change in childhood-onset systemic lupus eryth-
et al. Efficacy of cognitive behavioural therapy for the treatment of
ematosus. Arthritis Rheum 1999;42:1354–60.
chronic stress in patients with lupus erythematosus: a randomized con-
trolled trial. Psychother Psychosom 2010;79:107–15. 26. Procidano ME, Heller K. Measures of perceived social support from
friends and from family: three validation studies. Am J Community
14. Palermo TM, Wilson AC. eHealth applications in pediatric psychol-
Psychol 1983;11:1–24.
ogy. In: Roberts M, editor. Handbook of pediatric psychology
New York: Guilford; 2009. p.227–37. 27. Lyons JS, Perrotta P, Hancher-Kvam S. Perceived social support
from family and friends: measurement across disparate samples.
15. Tan EM, Cohen AS, Fries JF, et al. The 1982 revised criteria for the
J Pers Assess 1988;52:42–7.
classification of systemic lupus erythematosus. Arthritis Rheum 1982;
25:1271–7. 28. Rosenstiel AK, Keefe FJ. The use of coping strategies in chronic low
back pain patients: relationship to patient characteristics and current
16. Folstein MF, Folstein SE, McHugh PR. “Mini-mental state”. A prac- adjustment. Pain 1983;17:33–44.
tical method for grading the cognitive state of patients for the clinician.
J Psychiatr Res 1975;12:189–98. 29. Gil KM, Abrams MR, Phillips G, et al. Sickle cell disease pain:
relation of coping strategies to adjustment. J Consult Clin Psychol
17. Melzack R. The short-form McGill pain questionnaire. Pain 1987;30: 1989;57:725–31.
191–7.
30. Gil KM, Williams DA, Thompson RJ Jr, et al. Sickle cell disease in
18. Reynolds CR, Kamphaus R. Behavior assessment system for children. children and adolescents: the relation of child and parent pain coping
Circle Pines, MN: American Guidance; 1992. strategies to adjustment. J Pediatr Psychol 1991;16:643–63.
19. Merenda PF. BASC. Behavior assessment system for children. Meas 31. Turner JA, Romano JM. Psychological and psychosocial techniques: cog-
Eval Couns Dev 1996;28:229–332. nitive behavioral therapy. Pain Management [serial on the Internet] 1994.
20. Watson D, Clark L. The PANAS-X: preliminary manual for the 32. O’Donohue WT, Fisher JE. Adherence to pediatric treatment regi-
positive and negative affect schedule—expanded form. Dallas, TX: mens. In: O’Donohue M, Fisher JE, editors. General principles and
Department of Psychology, Southern Methodist University; 1991. empirically supported techniques of cognitive behavior therapy. Hobo-
21. Harter S. The self-perception profile for children: revision of the perceived ken, NJ: Wiley; 2009.
competence scale for children. Denver, CO: University of Denver; 1985. 33. Unnebrink K, Windeler J. Intention-to-treat: methods for dealing with
22. Thompson B, Butcher A, Berenson G. Children’s beliefs about sour- missing values in clinical trials of progressively deteriorating diseases.
ces of health: a reliability and validity study. Meas Eval Counsel Dev Stat Med 2001;20:3931–46.
1987;20:80–8. 34. O’Brien PC. Procedures for comparing samples with multiple end-
23. Varni JW, Seid M, Knight TS, et al. The PedsQLTM 4.0 Generic points. Biometrics 1984;40:1079–87.
Core Scales: sensitivity, responsiveness, and impact on clinical decision- 35. Schoenfeld D. Statistical considerations for pilot studies. Int J Radiat
making. J Behav Med 2002;25:175–93. Oncol Biol Phys 1980;6:371–4.

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