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Key words
spina bifida.
Introduction
Spina bifida is a birth defect caused by incomplete neural
tube development, resulting in a protrusion of spinal
cord, meninges, and nerve roots through an opening in
the spine (Burmeister et al., 2005). Most individuals with
spina bifida develop hydrocephalus, largely due to
herniation of the cerebellar tonsils through the foramen
magnum (i.e., ArnoldChiari II malformation). This
condition obstructs ventricular flow in the third and/or
fourth ventricles, leading to a posterior ! anterior
progression of ventricular dilation, and causing stretching
of nearby white matter tracts and compression of the
cortex (Bruner et al., 1999; Fletcher et al., 1996a;
Fletcher et al., 2000). Although children with spina bifida
tend to score within the low average to average range on
measures of general intellectual ability, they are at
heightened vulnerability to learning disabilities and
other cognitive difficulties (Brewer, Fletcher, Hiscock, &
Davidson, 2001; Fletcher et al., 1996b; Wills, 1993), and
tend to exhibit increased problems with socialization,
academic functioning, and vocational accomplishment as
they age (Dise, & Lohr, 1998; Holler, Fennel, Crosson,
All correspondence concerning this article should be addressed to Brigid Rose, VA Northern California Health
Care System (116), 150 Muir Road, Martinez, CA 94553. E-mail: brigid.rose2@va.gov.
Journal of Pediatric Psychology 32(8) pp. 983994, 2007
doi:10.1093/jpepsy/jsm042
Advance Access publication June 7, 2007
Journal of Pediatric Psychology vol. 32 no. 8 The Author 2007. Published by Oxford University Press on behalf of the Society of Pediatric Psychology.
All rights reserved. For permissions, please e-mail: journals.permissions@oxfordjournals.org
Objective This study was designed to examine attention processes and executive functioning in adolescents
with spina bifida, and to explore whether impairment in these domains contributes to problems with
social adjustment. Methods A sample of adolescents with spina bifida (n 68) and a matched comparison
group (n 68) and their families were followed longitudinally. All participants completed questionnaires,
and the adolescent participants underwent neurocognitive testing. Results The spina bifida sample
showed greater impairment on objective and subjective measures of attention and executive functioning,
even when differences in intellectual functioning were controlled. Additionally, attention and executive
deficits were found to be predictive of social adjustment difficulties. A mediational analysis suggested
the neurocognitive deficits mediate associations between spina bifida status and social adjustment
difficulties. Conclusions Adolescents with spina bifida appear to exhibit clear impairment in attention
and executive functioning and this impairment may contribute to their well-established social difficulties.
984
Method
Participants
The participants were drawn from a longitudinal study
focusing on family relationships and psychological
adjustment in children with spina bifida (Holmbeck
et al., 1997, 2002, & 2003). Children with spina bifida
between the ages of 8 and 9 were recruited for
Procedure
For each wave of data collection, trained research
assistants conducted home visits with the participating
families. During the visits, family members completed
questionnaires and participated in videotaped family
interaction tasks. Additionally, the child participants
were asked to participate in brief neurocognitive testing
(described subsequently) at Time 1 and Time 4.
Participating families were asked to sign release forms
allowing the research team to contact the childs teacher.
985
986
Measures: Examiner-Administered
Neuropsychological Instruments
Results
As noted above, 59 adolescents with spina bifida and 65
matched comparison adolescents were included in the
Time 4 analyses (n 124). At Time 1, the two samples
were found to differ significantly in estimated intellectual
Intellectual Functioning
At Time 1, each child was administered the Peabody
Picture Vocabulary Test, Revised (PPVT-R; Dunn &
Dunn, 1981), a test of receptive language that was used
as a proxy for general intellectual ability in the current
study. The test serves as a particularly useful estimate of
intellectual ability with the population under study
because its relatively rapid administration does not
penalize children who have difficulty sustaining attention
for long periods of time and it does not require fine
motor output like many other measures of intellectual
functioning. The test manual reports that the measure
correlates moderately well with the WISC and WISC-R
Full Scale IQ and Verbal IQ scores.
987
Focused Attention
Measure
Source
a
BRIEF Sustainb
BRIEF Working Memory
BRIEF Sustain
BRIEF Working Memory
Planning
Sample
BRIEF Organize
BRIEF Organize
Initiation
Initiation
BRIEF Initiate
BRIEF Initiate
Cohens d
6.06
3.10
50
1.37
Test data
Comparison
Spina Bifida
10.15
7.10
2.85
4.06
61
50
.98
Comparison
10.56
2.95
61
Spina Bifida
.75
.39
59
Comparison
.59
.38
64
Spina Bifida
.73
.47
59
Comparison
.48
.38
64
Teacher
Spina Bifida
.62
.47
48
.35
Teacher
Comparison
Spina Bifida
.46
.61
0.44
.57
62
48
.54
Comparison
.33
.46
62
Spina Bifida
6.27
Parents
Parents
Test data
Test data
Spina Bifida
10.18
6.02
10.21
5.90
3.15
49
3.11
62
3.18
50
2.77
62
.42
.58
1.25
1.41
Test data
Spina Bifida
3.23
50
1.45
Parents
Comparison
Spina Bifida
10.34
.76
2.87
.41
61
59
0.17
Comparison
.69
.41
64
Spina Bifida
.63
.56
48
Comparison
.43
.50
62
Spina Bifida
.84
.47
59
Comparison
.76
.45
64
Teacher
Spina Bifida
.54
.58
48
.56
Parents
Comparison
Spina Bifida
.27
.85
.37
.42
62
59
.50
Comparison
.65
0.38
64
Spina Bifida
.72
.57
48
Comparison
.47
.50
62
Teacher
Parents
Teacher
.38
.17
.46
Spina Bifida
Comparison
CAS Planned Connections
SD
Test data
Comparison
CAS Planned Codes
988
Preliminary Analyses
The groups were compared on all Time 4 social outcome
measures using a series of t-tests. Spina bifida participants
were rated as significantly more impaired on three of the
four variables: parent-reported SSRS item average score
(p .008), parent-reported SPPC Social Competence
score (p .016), and teacher-reported SPPC Social
Competence score (p .008). Differences were not
significant on the teacher SSRS.
Social Skills Rating Scale (SSRS)
Next, measures of attention and executive functioning
were tested as predictors of social impairment using
multiple regression (Table II). Group status (spina bifida
vs. comparison sample) and interactions between group
status and the neurocognitive predictors were added in
separate steps in the analyses to determine whether the
Table II. Multiple Regression Main Effects: Neuropsychological Predictors of Social Functioning
Dependent variable (source)
Predictor (source)
SSRS (parent)
Group
BRIEF mean (teacher)
SSRS (teacher)
.227
.040
4.43
.258
.060
6.82
.060
.050
.002
.002
0.23
.197
.308
.090
10.13
.231
.028
3.25
Group
.170
.022
2.27
R2
.049
.002
0.22
.348
.067
7.24
Group
.287
.064
6.74
.283
.294
.075
.046
8.59
5.49
Note: p <.05. p <.01. Beta weights are standardized and indicate the direction of the effect at the step the predictor entered the equation. The BRIEF is scored so that a
higher score indicates greater impairment, whereas on all other measures a higher score indicates better functioning. Thus, all main effects are in the expected direction.
None of the tested interaction effects were significant when entered separately, so these results were omitted from the table. PPVT-R scores were entered as a covariate in all
equations.
Exploratory Analyses
The results described above, specifically those that
showed an association between spina bifida and executive
impairment, and those that showed an association
between executive impairment and social difficulties,
lend some support to the theory offered by Landry
et al. (1993) and Fletcher et al. (1996b) that problems
with planning and other executive functions mediate
the relationship between spina bifida status and
social functioning (i.e., spina bifida status ! executive
impairment ! social difficulties). Thus, some exploratory
mediational analyses were included to test executive
functioning as a mediator of the well-established
link between spina bifida and social difficulties
(e.g., Holmbeck et al., 2003).
The preceding regression analyses (i.e., neurocognitive correlates of social adjustment) were run with and without PPVT-R
score as a covariate. The results changed minimally when PPVT-R
was in the equation. Therefore, it was not entered as a covariate in the
mediational analyses, as it would unnecessarily complicate the
analyses and interpretation. Because the CAS was found to be a
significant predictor of teacher-reported social competence only when
PPVT-R was controlled, this was not tested as a mediator.
989
990
Model One
.239** (.157ns ) z = 1.97*
.302** (.263**)
Executive
Functions
(Teacher BRIEF)
.257**
Group
Social Skills
(Parent SSRS)
Model Two
.217**(.032 ns) z = 2.33*
6.04**
Group
Executive
Functions
(CAS)
.303**(.284*)
Social Skills
(Parent Social
Competence)
.252**(.204*) z = 1.68ns
.188*
.188*
Group
Executive
Functions
(Parent BRIEF)
.306**(.261**)
Social Skills
(Teacher Social
Competence)
Discussion
The objectives of the current study were to identify the
aspects of higher order neurocognitive functioning most
affected by spina bifida and to understand the social
Model Three
991
992
Acknowledgment
Completion of this article was supported by Social and
Behavioral Sciences Research Grants from the March of
Dimes Birth Defects Foundation (#12-FY04-47) and a
grant from the National Institute of Child Health and
Human Development (NICHD; R01-HD048629). We
thank Ann Walsh Johnson, Joy Ito, Pat McGovern, Pat
Braun, Caroline Anderson, David McLone, and John
Lubicky; the staff of the spina bifida clinics at Childrens
Memorial Hospital, Shriners Hospital Chicago, and
Loyola University Chicago Medical Center; and the
Illinois Spina Bifida Association. We also thank numerous
undergraduate and graduate research assistants for help
with data collection and data entry. Most importantly, we
thank the parents, children, teachers, and health professionals who participated in this study.
Conflict of Interest: None declared.
Received May 11, 2006; revisions received April 14, 2007
accepted May 1, 2007
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