Hemophilia Growth and Development Study: Caregiver Report

of Youth and Family Adjustment to HIV Disease and

Immunologic Compromise
Janice D. Bordeaux,1 PhD, Katherine A. Loveland,2 PhD, David Lachar,2 PhD,
James Stehbens,3 PhD, Terece S. Bell,4 PhD, Sharon Nichols,5 PhD, Nancy Amodei,6 PhD,
Kristin Brelsford Adkins,7 BS, and the Hemophilia Growth and Development Study
1
Rice University; 2University of Texas Medical School, Houston; 3University of Iowa, College of
Medicine; 4Children’s Hospital of Los Angeles; 5University of California–San Diego Medical Center;
6
University of Texas Health Science Center, San Antonio; and 7University of Colorado, Boulder

Objective To assess differences in caregiver report of youth and family psychosocial adjust-

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ment associated with HIV infection and greater immune compromise in youths with hemo-
philia. Methods Caregivers of 162 boys with hemophilia 8 to 20 years old completed three
youth and family questionnaires (Personality Inventory for Children, Revised [PIC-R]; Ques-
tionnaire on Resources and Stress [QRS]; Family Environment Scale). Results Caregivers
of HIV positive (HIV+) youths reported greater health concerns, social withdrawal (PIC-R),
physical and adaptive limitations associated with illness (QRS) in their sons, and more pes-
simism about their sons’ future and negative attitudes about parenting (QRS). Caregivers of
HIV+ youths with greater immune compromise reported greater concerns about their sons’
health and greater pessimism about their futures, as well as lower levels of family integration
and more limited family opportunities. Conclusions Results suggest caregivers perceive
psychosocial problems in HIV+ youths with hemophilia and their families; some problems are
specifically associated with greater immune compromise.

Key words hemophilia; HIV; psychosocial; youth; family stress; immune compromise.

Youths with hemophilia and HIV infection form a unique
cohort for studying the long-term developmental effects of
HIV infection. Most were infected past infancy, and most
lack the risk factors associated with vertical transmission,
including lower socioeconomic status (SES) and mater-
nal HIV infection. The Hemophilia Growth and Develop-
ment Study (HGDS), a multicenter, multidisciplinary lon-
gitudinal study, was formed to follow the growth and
development of these young people (Loveland et al., 1994,
2000; Nichols et al., 2000; Stehbens et al., 1997). In this
article we report results from the Family Stress and Cop-
ing Study (FSCS), an HGDS adjunct study on caregiver
report of the psychosocial adjustment and family func-
tioning of 162 male youths with hemophilia ages 8 to 20
with and without HIV infection.
The psychosocial adjustment of youths with HIV in-
fection and their families may be affected by the cogni-
tive effects, social stigma, and life-threatening nature of
HIV infection (Kazak, 1997; Lewis, 2001; Moss, Bose,
Wolters, & Brouwers, 1998). Some studies suggest that
youths with HIV exhibit more anxiety disorders (Bussing
& Burket, 1993) and depression (Burton, Sarkis, Hill,
Kemph, & Mehta, 1991). A longitudinal study examin-
ing youth and parent report of adjustment in children and
adolescents with HIV (both vertical transmission and
transfusion associated) found that youths self-reported

All correspondence should be sent to Janice D. Bordeaux, Office of the Dean, George R. Brown School of Engineering,
MS-364, Rice University, 6100 S. Main St., Houston, Texas. E-mail: jbordeau@rice.edu. Anne Kazak, PhD, ABPP,
former Editor, served as accepting editor on this article.

Journal of Pediatric Psychology, Vol.  No. , , pp. – ©  Society of Pediatric Psychology
DOI: ./jpepsy/jsg
 Bordeaux et al.
elevated depression and anxiety (Bose, Moss, Brouwers,
Pizzo, & Lorion, 1994; Moss et al., 1998). Parents reported
that their sons had problems with social functioning, anx-
iety, and conduct that persisted at a 2-year follow up.
Bachanas, Kullgren, Schwartz, Lanier, et al. (2001) found
that parents reported greater emotional and behavioral
problems for youths with vertically transmitted HIV in-
fection than expected for their ages but no more adjust-
ment problems than a healthy comparison group of similar
SES. This result could reflect availability of psychosocial
services for the group with HIV, or underreporting of prob-
lems by parents. Similarly, Nichols et al. (2000) reported
that HGDS youths with HIV and hemophilia had fewer
externalizing behavior problems over time, suggesting a de-
crease due to advancing illness, or a shift in parents’ focus
from their son’s behavior to his health problems. How-
ever, other findings from the HGDS (Loveland et al., 2000;
Nichols et al.) indicate that significant declines in im-
mune functioning in youths with hemophilia and HIV are
associated with declines in cognitive and adaptive func-
tioning, which may in turn affect emotional and behavioral
functioning. A large study of youths with hemophilia
(Drotar, Agle, Eckl, & Thompson, 1997) found no differ-
ences in psychosocial adjustment between those with and
without HIV, although it excluded youths with HIV-related
immune compromise and dementia, a subgroup likely
to be more at risk. These findings suggest that effects of
chronic HIV on youth adjustment may differ by associ-
ated risk factors, illness course, and availability of services.
The effects of HIV disease on youths should also be
considered within the context of family adjustment (Dro-
tar, 1997). In addition to potential stressors associated
with chronic illness, such as increased financial burden,
medical procedures, disruption of normal activities, and
less equitable relations with healthy siblings (Hamlett,
Pellegrini, & Katz, 1992; Holden, Chmielewski, Nelson,
Kager, & Foltz, 1997; Kazak, 1989; Lewis, Haiken, &
Hoyt, 1994), HIV disease can be associated with fear of os-
tracism, severe debilitation, premature death, and even
deaths across generations (Bose et al., 1994; Kazak, 1997;
Mellins & Ehrhardt, 1994; Sherwen & Boland, 1994;
Wiener, Vasquez, & Battles, 2001). Drotar et al. (1997)
found that compared to mothers of uninfected youths
with hemophilia, mothers of youths with both hemophilia
and HIV reported a greater impact of negative life events
on mothers’ psychological distress, suggesting that HIV
disease may increase the family’s vulnerability to stress.
Although Bachanas, Kullgren, Schwartz, McDaniel, et al.
(2001) found that caregivers of vertically infected youths
did not differ in distress and adjustment problems from
caregivers of youths without HIV, this finding could re-
flect the presence of additional risk factors (e.g., lower
SES) whose effects might outweigh effects of HIV.
The illness course of HIV, together with both stres-
sors and resources for coping, can affect both youth ad-
justment and family functioning. Chronic HIV disease in
young men with hemophilia may have a prolonged asymp-
tomatic phase that may be less stressful for youths and
their families than the later stages of HIV, which are
marked by immune compromise (Hilgartner et al., 1993;
Loveland et al., 1994). Declines in cognitive and adaptive
functioning associated with advancing immune compro-
mise and illness (Loveland et al., 2000; Nichols et al.,
2000) could severely stress the family and reduce resources
for coping, leading to poorer outcomes. Moss et al. (1998)
found that, in children with HIV, negative life events were
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associated with poorer psychosocial outcomes, but that
psychological resilience to stress had a stabilizing effect.
Five children with HIV who died within 1 year of baseline
were found to have more negative life events and less re-
silience to stress, as well as greater progression of HIV
disease. Taken together, these findings suggest that psy-
chosocial adjustment and family functioning in youths
with hemophilia may be adversely affected both by the
presence of HIV infection and by the lowered immune
functioning that characterizes later stages of HIV disease.
The purpose of this study was to examine the rela-
tionship of youth and family psychosocial functioning to
both HIV serostatus and the extent of immune compromise
in male youths with hemophilia. This hemophilia cohort
was stratified into two groups based on youth HIV sero-
status: a group with HIV (HIV+; n = 86) and a group with
no HIV (HIV–; n = 76). The HIV+ group was further di-
vided into subgroups based on degree of immune dys-
function: (a) HIV+ with CD4+ t-cell counts (CD4 cells/
mm3) < 200 (HIV+ group with lower immunity; n = 37),
and (b) HIV+ with CD4+ t-cell counts ≥ 200 (HIV+ group
with higher immunity; n = 49). Caregivers completed
three standardized instruments measuring youth psy-
chosocial adjustment, family resources and stress, and
family social environment. Caregiver-report only was ob-
tained for this study because of the already high burden of
testing placed on youth participants in the HGDS. Con-
current measures of youths’ HIV status, immune status,
and intelligence and family demographic characteristics
were available from the HGDS; these variables have been
described earlier in detail (Loveland et al., 1994; Stehbens
et al., 1997). They are summarized in Table I.
The primary research questions of this study were
the following. Do youth and family psychosocial prob-
lems differ between families whose sons have both he-
mophilia and HIV infection and those whose sons have

Table I. Demographic Characteristics of Youths With Hemophilia, With and Without HIV Infection
All HIV—
(n = 76)
Characteristics M (SD)
Age (yrs) 13.3a (3.0)
Full-Scale IQ (WISC-R) 108.1 (17.2)
Mother’s yrs of school 12.6 (2.5)
Father’s yrs of school 12.9 (3.1)
Absolute CD4 cells/mm3 827.3b (294.6)
a
F (1, 161) = 9.3, p = .003.
b
F (1, 161) = 133.4, p = .000.
c
F (1, 161) = 130.7, p = .000.
hemophilia alone? Are greater youth and family psy-
chosocial problems associated with immune compromise
in youths with hemophilia and HIV infection? Hypothesis
1: Families with sons who have both hemophilia and HIV
infection will have greater psychosocial problems than
families with sons who have hemophilia alone, as mea-
sured by youth psychological adjustment, family resources
and stress, and family social environment. Hypothesis 2:
Severe immune compromise in sons with hemophilia and
HIV will be associated with greater psychosocial prob-
lems, as measured by youth psychological adjustment, fam-
ily resources and stress, and family social environment.
Method
Participants
FSCS participants were the caregivers or legal guardians of
162 youths with hemophilia ages 8 to 20 (M = 14.0 years,
SD = 3.0). Respondent caregivers were almost all mothers,
with 3% fathers and 4% other. All were the primary care-
giver. Characteristics of the caregivers and youths are
given in Table I, including youth age, full-scale IQ, and
CD4 cell count, along with mean years of education for
mothers and fathers.
Of 14 HGDS centers, 10 enrolled caregivers in the
FSCS. Not all eligible centers chose to participate, because
of the time and training required for professional staff.
FSCS participants represented 63% of all HGDS caregivers
and 81% of HGDS caregivers at the participating FSCS
sites. Eleven caregivers (13.6%) had two sons enrolled;
10 of them had both sons in the HIV– group. The eleventh
caregiver’s sons were both HIV+, and both had CD4 cell
counts ≥ 200.
Family structure was coded in the HGDS baseline
history. At enrollment, 78% were two-parent families,
20.4% were single-mother-headed families, and the re-
mainder were single-father-headed or other (grandparent
or other legal guardian). SES was estimated using the Re-
Youth and Family Adjustment 
All HIV+ HIV+,CD4 <200 HIV+,CD4 ≥200
(n = 86) (n = 37) (n = 49)
M (SD) M (SD) M (SD)
14.7a (2.9) 14.8 (2.9) 14.7 (2.9)
105.4 (15.3) 106.9 (16.2) 104.3 (14.7)
12.6 (3.0) 13.1 (2.8) 12.2 (3.1)
12.5 (3.5) 12.9 (3.9) 12.2 (3.2)
301.5b (286.1) 40.0c (48.3) 489.8c (232.2)
vised Socioeconomic Index of Occupational Status (SEI),
with possible scores from 13.8 to 90.4 (Stevens & Feath-
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erman, 1981; see Stehbens et al., 1997, for discussion of
methods). Families in the FSCS had an average SEI of
34.2 (SD = 18.5) (middle class). On average, respondent
caregivers had completed high school (M = 12.6 years of
education, SD = 2.7). Neither the SEI nor the caregiver
education values differed significantly from those of the
HGDS full sample (Stehbens et al., 1997).
Study measures and HGDS medical and neuropsy-
chological evaluations were obtained between March 1992
and November 1993, on average about 10 years since in-
fection (see Loveland et al., 1994, 2000, for discussion of
how time of infection was estimated). For all but six fam-
ilies, data were collected at the third annual follow-up
assessment past baseline, and for the other six families,
at the fourth annual follow-up. Following institutional
review board approval, caregivers gave informed consent.
Informed consent/assent by sons was required for care-
giver participation.
FSCS youths were representative of boys with hemo-
philia in the United States who were at risk for acquiring
HIV disease through contaminated blood clotting factor
products before 1985: 53% were HIV+, 67% Caucasian,
17% African American, and 16% Hispanic (Daar et al.,
2001). For the purpose of analysis, ethnic status was classi-
fied into majority (Caucasian) and minority (African Amer-
ican, Hispanic, Latino, and Chicano) groups. No families
of Asian ethnicities volunteered to enroll in the FSCS.
The HGDS sample (n = 333) was representative of
the total population of boys with moderate to severe he-
mophilia at the 14 HGDS centers (Hilgartner et al., 1993).
Because the FSCS respondents were a self-selected sub-
set of HGDS caregivers, analyses were conducted using t
tests and the chi-square statistic to compare characteristics
of the HGDS participants at baseline who were or were
not enrolled in the FSCS. FSCS youths were significantly
younger (M = 14.0 vs. 15.6 years, SD = 2.97 and 3.40, re-
 Bordeaux et al.
spectively), t(332) = 3.70, p < .0003, than those in the
HGDS full sample. This difference may reflect the fact
that many older participants came to HGDS study visits
without their parents, reducing the opportunity for par-
ticipation in this study. Based on CD4+ t-cell counts,
youths in the FSCS were of similar immune status to
youths whose caregivers did not participate (22.8% FSCS
vs. 22.2% HGDS with CD4+ cells/mm3 < 200). No signif-
icant differences were found in type of hemophilia diag-
nosed (A or B), ethnic status, youth intelligence (Wechsler
Intelligence Scale for Children-Revised or Wechsler Adult
Intelligence Scale-Revised) or social maturity (Vineland
Adaptive Behavior Scales Composite Score).
Procedure
Study measures were selected based upon empirical evi-
dence validating their use in measuring youth psychoso-
cial adjustment, family stress and resources, and family
environment.
Personality Inventory for Children-Revised (PIC-R; Lachar,
1982; Lachar & Kline, 1994). This empirically derived
280-item true/false instrument (caregiver report) assesses
psychosocial adjustment in preschool through adolescent
youths. Caregivers completed the PIC-R with reference
to their child who was enrolled in the FSCS. Twelve scales
measure three development dimensions (achievement,
development, intelligence) and nine adjustment dimen-
sions (anxiety, depression, delinquency, family relations,
hyperactivity, psychosis, social skills, somatic concern,
withdrawal). The scales are interpreted through actuar-
ial guidelines derived for T-score ranges that vary by scale
(Lachar & Gdowski, 1979).
Questionnaire on Resources and Stress-Long Form (QRS;
Holroyd 1974, 1987). This 285-item true/false caregiver
questionnaire with 15 scales measures the impact of child-
hood chronic illness or disabilities on stress in the care-
giver, family, and youths. Caregivers completed this scale
with reference to the youths enrolled in the FSCS and the
family. Caregiver scales include Poor Health/Mood (role
discontent and sadness), Excess Time Demands, Nega-
tive Attitude (about youths), Overprotective/ Dependency
(of youths), Lack of Social Support, Overcommitment/
Martyrdom, and Pessimism (about the youth’s future).
Family stress scales are Lack of Family Integration, Fi-
nancial Problems, and Limits on Family Opportunity.
Scales describing the youths with illness (Index Case Prob-
lems) are Physical Incapacitation, Difficult Personality
Characteristics, Social Obtrusiveness, Limited Activities,
and Occupational Limitations for Index Case.
Family Environment Scale (FES; Moos & Moos, 1981).
This 90-item true/false questionnaire measures three di-
mensions: family relationships (family cohesion, conflict,
and expressiveness), emphasis on types of personal growth
(achievement orientation, independence, intellectual-
cultural orientation, active-recreational orientation, and
moral-religious emphasis), and family system mainte-
nance (organization and control). Caregivers completed
this scale with reference to the family as a whole.
Statistical Methods
Multivariate and univariate analyses of variance
(MANOVA, ANOVA) were used to evaluate the two
hypothesis-relevant group comparisons (HIV+/HIV– and
greater/lesser immune compromise) across each of the
three sets of scales provided by the PIC-R, QRS, and FES.
Because age differed significantly between participants
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with and without HIV infection, age was entered as a co-
variate in the parametric comparisons conducted by HIV
status. Because of the unique, nonreplicable nature of
these data, an exploratory approach was applied to data
analysis. ANOVAs of individual scale dimensions were
conducted regardless of the statistical significance of the
associated MANOVA. However, caution was introduced
into these analyses through the additional estimation of the
magnitude and pragmatic value of all statistically sig-
nificant ANOVAs by the application of two other pro-
cedures. The magnitude of statistically significant uni-
variate group differences was assessed through the
calculation of a standardized effect size (d), in which val-
ues reflect either small (d = .20), medium (d = .50), or
large (d = .80) effects (Cohen, 1988). A second procedure
estimated the practical value of statistically significant
group differences through the classification of individual
subjects. Chi-square was applied to evaluate differences
in proportions of clinically elevated scale T-scores. For
the PIC-R, clinical elevations were defined empirically
by Lachar and Gdowski (1979) for individual scales, as
either T > 59, T > 69, or T > 79. For the QRS and FES,
for which comparable actuarial guidelines were not avail-
able, a liberal criterion of one standard deviation from the
normative mean was used, as presented in the measure’s
manual (T > 59). Because the HIV– group included 11
sibling pairs, we also repeated the parametric analyses
with these 22 participants removed. Results were the same
as those obtained from analyses of the full sample.
Results
Comparison of HIV+ and HIV– Samples
The three sets of caregiver scales were first analyzed to
evaluate the caregivers’ perception of the effect of HIV on
their sons with hemophilia. PIC-R scales differed signifi-
 Youth and Family Adjustment 

Table II. HIV Status Differences in Caregiver Descriptions of Youths With Hemophilia
HIV– HIV+

M (SD) % M (SD) % F d χ2

Personality Inventory for Children, Revised

(HIV– n = 74; HIV+ n = 80)
Somatic Concern (T > 69) 56.9 (13.2) 15 64.2 (13.9) 40 9.65** .54 12.1**
Depression (T > 69) 54.0 (13.1) 16 59.4 (14.2) 25 7.28** .39 1.8
Withdrawal (T > 69) 53.4 (12.1) 9 59.7 (15.0) 24 8.90** .46 5.6*
Anxiety (T > 69) 53.2 (10.9) 7 55.5 (12.4) 14 4.54* .20 2.0
Social Skills (T > 69) 46.9 (10.7) 0 48.9 (11.6) 3 3.90* .18 1.9
Questionnaire on Resources and Stress: Parent Problems
(HIV– n = 74; HIV+ n = 81)
Poor Health/Mood (T > 59) 3.2 (2.9) 43 4.8 (3.1) 59 13.12** .53 1.5
Negative Attitude About Youth (T > 59) 5.5 (2.4) 32 7.1 (2.9) 54 17.64** .60 7.5**
Overprotection/Dependency (T > 59) 3.6 (2.3) 26 4.9 (2.6) 38 14.59** .53 2.8
Pessimism About Youth (T > 59) 2.7 (1.5) 34 4.7 (2.1) 68 35.89** 1.09 18.0**

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Questionnaire on Resources and Stress: Family
Functioning Problems
Lack of Family Integration (T > 59) 3.0 (2.4) 24 4.0 (2.8) 38 4.78* .38 3.5
Limits on Family Opportunity (T >59) 1.2 (1.8) 26 1.8 (1.9) 41 4.71* .32 3.9
Questionnaire on Resources and Stress: Index Case Problems
Physical Incapacitation (T > 59) 2.4 (1.3) 47 3.1 (1.6) 70 7.66** .48 6.7**
Limited Activities (T > 59) 0.8 (1.3) 20 1.5 (1.4) 43 12.56** .52 9.3**
Difficult Personality Characteristics (T > 59) 4.3 (4.2) 27 5.3 (4.2) 33 4.58* .24 0.7
Family Environment Scale: Personal Growth Dimension
(HIV–n = 75; HIV+ n = 83)
Independence (T > 59) 45.7 (11.2) 16 50.9 (12.1) 30 4.79* .45 4.4*
Detail for significant ANOVAs only; T values in parentheses are clinical elevations.
% = proportion in clinical range, F = difference in group means adjusted for age, d = standardized effect, χ2 = difference in elevated score group proportions.
*≤ .05.
**≤ .01.

cantly by HIV status when age was entered as a covariate
(Wilks λ = .852), F(12, 140) = 2.02, p = .03. Significant
(p < .05) ANOVAs were obtained for 5 of 12 PIC-R sub-
stantive profile scale T-score values (Table II). Youths with
HIV were described as having more evidence of compro-
mised health (Somatic Concern) and relative isolation
(Withdrawal); these differences represented a medium
effect. Significant differences were also found for PIC-R
scales reflecting dysphoria (Depression), tension and worry
(Anxiety), and poor peer relations (Social Skills), although
they represented only a small effect. When frequency of
clinically elevated scores was examined for these five
scales, two PIC-R scales differed significantly between
groups, although neither was descriptive of the majority of
HIV+ youths (40% of HIV+ youths had an elevated Somatic
Concern scale, while only 24% had an elevated With-
drawal scale).
QRS scales differed significantly by HIV status when
age was entered as a covariate (Wilks λ = .725), F(15,
138) = 3.14, p = .0001. Significant (p < .05) ANOVAs
were obtained for 9 of 15 scales (Table II). Of the seven
scales that describe the caregiver’s adjustment, experience,
and attitudes, four differed significantly by HIV status and
represented at least a medium effect. Caregivers of HIV+
youths were more likely to describe themselves as more
distressed (Poor Health/Mood), fearful about their son’s
future (Pessimism), concerned over the amount of care
necessary for their son (Overprotective/Dependency), and
having negative parenting attitudes (Negative Attitude)
compared to caregivers of HIV– youths. Although the
QRS manual provides norms based only on a sample of
52 caregivers, scale scores equivalent to T = 60 are given.
Classification of QRS scale scores based on these values
resulted in a significantly higher proportion of elevated
values for the caregivers of HIV+ youths for two of these
scales, Pessimism (68%) and Negative Attitude (54%).
Two of three QRS scales that describe family status
(Limits on Family Opportunity and Lack of Family Inte-
gration) differed significantly by HIV status, although
these differences represented a small effect and resulted in
 Bordeaux et al.

Table III. CD4 Status Differences in Caregiver Descriptions of HIV+ Youths With Hemophilia
CD4 > 200 (n = 47) CD4 < 200 (n = 33)

M (SD) % M (SD) % F d χ2

Personality Inventory for Children, Revised

Somatic Concern (T > 69) 60.2 (13.3) 26 69.4 (13.3) 61 8.07** .69 9.9**
Questionnaire on Resources and Stress: Parent Problems
Pessimism About Youth (T > 59) 4.1 (2.0) 57 5.4 (2.1) 82 6.10* .64 5.6*
Questionnaire on Resources and Stress: Family Functioning Problems
Lack of Family Integration (T > 59) 3.3 (2.5) 26 5.0 (2.9) 56 7.36** .64 7.7**
Limits on Family Opportunity (T > 59) 1.4 (1.7) 34 2.3 (2.0) 50 4.85* .49 2.1
Detail provided only for significant scale ANOVAs; T value in parentheses designates clinical elevations.
% = proportion in clinical range, F = differences in group means, d = standardized effect size, χ2 = difference in % elevated scores.
*≤ .05.
**≤ .01

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no difference in proportion of cases classified within the
clinical range. Three of five QRS scales describing study
youths differed significantly by HIV status, and two of
these obtained a group difference that represented a
medium effect (Physical Incapacitation and Limited
Activities). The QRS measure of poor personal adjust-
ment (Difficult Personality Characteristics) also sug-
gested that HIV infection represents additional threat to
youth adjustment over hemophilia alone, although this
scale obtained only a small effect and did not obtain a
greater proportion of clinically elevated scores in HIV+
youths.
MANOVA of the FES did not generate a statistically
significant effect for HIV status when age was entered
as a covariate (Wilks λ = .905), F(10, 146) = 1.54, p =
.13, although 1 of 10 scales (Independence) obtained a
statistically significant ANOVA by HIV status (Table II).
The families of HIV+ sons were described as more likely to
promote personal growth through an emphasis on inde-
pendence.
range, 61% of the more immune compromised youths had
an elevated Somatic Concern value compared to only 26%
of the remaining youths (Table III).
The QRS statistically differed by degree of HIV im-
mune compromise (Wilks λ = .677), F(15, 65) = 2.07, p =
.02, although only 3 of the 15 QRS scales obtained a sig-
nificant univariate group effect; each of these differences
represented a medium effect. As expected, the majority
(82%) of caregivers were pessimistic about the future for
HIV+ sons with CD4 values < 200 (Pessimism). Two QRS
measures of family environment also differed by presence
of immune compromise (Lack of Family Integration and
Limits on Family Opportunity), representing a poorer
family adjustment with deteriorating immune status in
the HIV+ son.
MANOVA of the FES did not result in a statistically
significant effect of HIV+ immune compromise (n = 34,
CD4 < 200 vs. n = 47, CD4 > 200, Wilks λ = .863), F(10,
72) = 1.14, p = .34. Similarly, all 10 of the subsequent uni-
variate FES comparisons were not significant.

Comparison of HIV+ Samples of High and Low

Immune Compromise
Comparable analyses of caregiver scales were conducted
within the sample to compare youths with substantial im-
mune compromise (CD4 < 200, n = 33) with the remain-
der of the HIV+ sample (n = 47). MANOVA of the 12 PIC-
R scales was not significant for the main effect of HIV+
immune compromise (Wilks λ = .784), F(12, 67) = 1.53,
p = .13; only one scale obtained a statistically significant
univariate group difference, although the difference was
considerable (d = .69). HIV+ youths with significant im-
mune compromise had higher average scores on Somatic
Concern than HIV+ youths with an adequate CD4 count.
When individual youths were classified within the clinical
Discussion
We hypothesized that caregivers of youths with hemo-
philia and HIV would report greater psychosocial prob-
lems for both the son and family compared to caregivers
of sons with hemophilia alone. The groups differed on
measures of youth psychological adjustment, particularly
health concerns and social withdrawal (PIC-R) and adap-
tive limitations associated with illness (QRS). They also dif-
fered on measures of the caregiver’s adjustment to the
youth’s illness, particularly pessimism about the son’s
future and negative attitudes about parenting (QRS). The
groups did not differ on most aspects of family social en-
vironment. These results support previous studies that

have suggested that youths with both hemophilia and HIV
and their families are at greater risk for psychosocial prob-
lems than those youths and their families with hemophilia
alone (Drotar et al., 1997; Nichols et al., 2000). Our results
also clearly indicate concerns by parents of HIV+ youths
regarding effects of advancing illness associated with HIV,
beyond those concerns related to hemophilia itself.
Greater immune compromise (CD4 cells/mm3 < 200)
in sons with HIV was hypothesized to be associated with
differences in youth adjustment and family stress and cop-
ing resources. Caregivers of HIV+ youths with greater im-
mune compromise did report significantly greater con-
cerns about health, and since severe immune deficiency is
associated with a late stage of illness, it is not surprising
that 82% of caregivers with immune compromised sons
also expressed greater pessimism about their sons’ future.
The fact that a greater proportion of these same caregivers
reported lower levels of family integration may suggest
that stress related to the son’s advancing illness may ad-
versely affect relationships within the family. These re-
sults indicate that families and youths were aware of health
risks and that their concerns were realistically linked to the
presence and progress of HIV disease.
The lack of differences in family environment found
between groups with and without HIV and with and with-
out significant immune compromise suggests that differ-
ences in caregiver perceptions of their sons were prob-
ably not due to differences in family relationships,
organization, or activities they value. Rather, these findings
are consistent with research that suggests that negative
life events and advancing HIV illness are important de-
terminants of family stress and psychosocial adjustment in
youths with HIV (Drotar et al., 1997). These findings also
emphasize that future studies must assess both healthy
and medically compromised youth to capture the psy-
chosocial impact of the full HIV disease spectrum.
Conclusions from this study are limited by the fact
that results are based only on caregiver report. It is rea-
sonable to expect that youths with HIV disease might view
their own behavior and emotional state, as well as family
functioning, somewhat differently than would their parents
(Drotar et al., 1996.) Thus, our findings reflect caregivers’
perception of the extent of youth and family problems as-
sociated with HIV infection and advancing disease course.
Because of their day-to-day role in managing the medical
care of their sons with hemophilia, these caregivers, largely
mothers, may have a perspective on youth and family
problems that differs both from that of the youths them-
selves and from that of fathers. Also, some research sug-
gests that caregivers who are themselves distressed may be
more likely to report distress or other psychosocial prob-
Youth and Family Adjustment 
lems in their sons with chronic illness (Bachanas, Kull-
gren, Schwartz, McDaniel, et al., 2001; Chilcoat & Breslau,
1997; Renouf & Kovacs, 1994.) Thus, future studies
should include both self-report and caregiver-report mea-
sures to address the perceptions of youths with HIV of
their own adjustment and family functioning, as well as
differences between self- and parent-report.
These findings indicate that caregivers’ perceptions
of the psychosocial functioning of youths with HIV and
their families, including the functioning of the caregivers
themselves, are related to the youth’s degree of illness.
Coping strategies focused on problem solving rather than
emotions have been associated with better psychosocial
outcome among youths with HIV and their caregivers
(Bachanas, Kullgren, Schwartz, Lanier, et al., 2001;
Downloaded from jpepsy.oxfordjournals.org by guest on February 1, 2011
Bachanas, Kullgren, Schwartz, McDaniel, et al., 2001).
Thus, psychosocial interventions to improve coping with
HIV illness and the negative events associated with disease
progress may be a critical need for families of HIV+ youths
with hemophilia.
Appendix
HGDS Center Directors, Study Coordinators or
Committee Chairs
Children’s Hospital Los Angeles: E. Gomperts, MD, W. Y.
Wong, MD, F. Kaufman, MD, M. Nelson, MD, S. Pearson,
RN; University of Texas Medical School, Houston: W. K.
Hoots, MD, K. Loveland, PhD, M. Cantini, RN; The Na-
tional Institutes of Health, National Institute of Child
Health and Human Development: A. Willoughby, MD,
MPH, R. Nugent, PhD; New England Research Institutes,
Inc.: S. McKinlay, PhD; Rho, Inc.: S. Donfield, PhD; Uni- AQ3
versity of Iowa Hospitals and Clinics: C. T. Kisker, MD,
J. Stehbens, PhD, S. O’Conner, J. McKillip, RN; Tulane
University Medical Center: P. Sirois, PhD; Children’s Hos-
pital of Oklahoma: C. Sexauer, MD, H. Huszti, PhD,
F. Kiplinger, S. Hawk, PA-C.; University of Texas Health
Science Center, San Antonio: J. Mangos, MD, A. Scott,
PhD, R. Davis, RN; Milton S. Hershey Medical Center:
M. E. Eyster, MD, D. Ungar, MD, S. Neagley, RN, MA; Uni-
versity of California-San Diego Medical Center: G. Davi-
gnon, MD, P. Mollen, RN; Kansas City School of Medi-
cine, Children’s Mercy Hospital: B. Wicklund, MD, A.
Mehrhof, RN, MSN.
Acknowledgments
We are grateful to the HGDS families who contributed
their invaluable time and effort to this project. We also
thank Sharyne Donfield, Margaret Maeder, and the New
 Bordeaux et al.
England Research Institute for their outstanding admin-
istrative support, without which the study could not have
been accomplished, and Charles Contant, Jr., for his help
in data management and analysis. This work was sup-
ported by the Bureau of Maternal and Child Health and
Resources Development (MCJ-060570), the NICHD (NO1-
HD-4 –3200), the Centers for Disease Control and Pre-
vention, and NIMH. Additional support was provided by
grants from the National Center for Research Resources of
the NIH to the New York Hospital Cornell Medical Clin-
ical Research Center (MO1-RR06020), the Mount Sinai
General Clinical Research Center, New York (MO1-
RR00071), the University of Iowa Clinical Research Cen-
ter (MO1-RR00059), and the University of Texas Health
Science Center, Houston (MO1-RR02558).
Received January 3, 2001; revisions received October 10,
2001, and February 6, 2002; accepted June 3, 2002
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