A multimethod,multi-informant,and multidimensional perspective on psychosocial adjustment in preadolescents with spina bifida

This study examined the psychosocial adjustment of preadolescents with spina bifida in relation to a comparison sample of able-bodied preadolescents (8- and 9-year-olds; n = 68 in each sample). The study also examined the potential clinical utility of a narrowband multimethod, multi-informant, and multidimensional perspective on the assessment of psychosocial functioning in children and adolescents with pediatric conditions. Findings revealed that children with spina bifida tended to be socially immature and passive, less likely to have social contacts outside of school, more dependent on adults for guidance, less competent scholastically, less physically active, less likely to make independent decisions, and more likely to exhibit attention and concentration difficulties. No group differences were found for externalizing symptoms, affective functioning, or global self-worth, suggesting resilience in these domains for the spina bifida sample. Findings also suggest that low socioeconomic status and the presence of a physical disability may be additive risk factors for certain psychosocial adjustment difficulties.     

Condition severity and psychosocial functioning in pre-adolescents with spina bifida: disentangling proximal functional status and distal adjustment outcomes

OBJECTIVE: To examine relations between condition severity and psychosocial functioning in 70 8- and 9-year-old pre-adolescents with spina bifida by testing several direct, indirect, and mediated effects models for proximal functional status and distal adjustment outcomes. METHODS: Proximal functional status outcomes (e.g., degree of involvement in activities, scholastic competence, athletic competence, attentional problems) and distal adjustment outcomes (e.g., behavior problems, social competence) were assessed with mother, father, and teacher report. Severity variables included spinal lesion level, spina bifida classification, shunt status, ambulation status, number of shunt surgeries, and two severity composites. RESULTS: Condition severity was associated with the proximal functional status outcomes across parent and teacher report. In contrast, no significant relationships were found between the severity parameters and distal adjustment outcomes. Findings supported a proximal effects model of condition severity as well as an indirect effects model (e.g., presence of a shunt-->less scholastic competence-->less social competence) and were consistent with recent theoretical formulations (e.g., Wallander & Varni, 1995). CONCLUSIONS: Disentangling proximal functional status outcomes and distal adjustment outcomes is critical in studies of condition severity and psychosocial functioning. We discuss clinical implications.     

Children With Chronic Physical Disorders: Maternal Reports of Their Psychological Adjustment

Advances in biomedical science have resulted in dramatic improvementsin the medical care of chronically ill and handicapped children.Past measurement problems have resulted in a lack of clarityregarding the psychological adjustment of these children. Themothers of 270 chronically ill and handicapped children wereadministered the Child Behavior Checklist in an attempt to identifypatterns of behavioral functioning across six pediatric chronicdisorders: juvenile diabetes, spina bifida, hemophilia, chronicobesity, juvenile rheumatoid arthritis, and cerebral palsy.In general, it was found that children in all chronic disordergroups were perceived by their mothers as evidencing on theaverage more behavioral and social competence problems thanexpected based on norms for children in general. However, theirbehavioral and social adjustment was reported as better thanthat of a normative sample of children referred to mental healthclinics. There were essentially no differences between childrenwith different chronic disorders in terms of behavior problemsand social competence. The results were taken to support theview that these children were as a group at risk for adjustmentproblems. They were also discussed in terms of the noncategorialapproach, which suggests that similar psychosocial challengesare faced across pediatric chronic physical disorders.     

Observed and perceived parental overprotection in relation to psychosocial adjustment in preadolescents with a physical disability: the mediational role of behavioral autonomy

The purpose of this study was to tes a mediational model of associations between parental overprotectiveness (OP), behavioral autonomy. and psychosocial adjustment in 68 families with 8- and 9-year-old preadolescents with spipa bifida and a demographically matched sample of 68 families with able-bodied children. Measures included questionnaire and observational assessments of parental OP; parent and child reports of behavioral autonomy; and parent, child, and teacher reports of preadolescent adjustment. On the basis of both questionnaire and observational measures of OP, mothers and fathers of children with spina bifida were significantly more overprotective than their counterparts in the able-bodied sample, although this group difference was partially mediated by children's cognitive ability. Across samples, mothers were more likely to be overprotective than fathers. Both questionnaire and observational measures of parental OP were associated with lower levels of preadolescent decision-making autonomy as well as with parents being less willing to grant autonomy to their offspring in the future. For the questionnaire measure of OP, and only for the spina bifida sample. the mediational model was supported such that parental OP was associated with less behavioral autonomy, which was, in turn, associated with more externalizing problems. Findings are discussed in relation to the literature on parenting, autonomy development, and pediatric psychology.     

Maternal, Paternal, and Marital Functioning in Families of Preadolescents with Spina Bifida

Based on a family systems/social-ecological perspective, mothersand fathers of 8-and 9-year-old children with spina bifida (n=55;28male, 27 female) were examined in comparison to a matched groupof parents with 8-and 9-year-old able-bodied children (n=55;29 male, 26 female) across several areas of functioning (individual,parental and marital). Findings suggested that mothers and fathersin the spina bifida sample tended to report more psychosocialstress than their counterparts in the able-bodied sample. Specifically,mothers and fathers in the spina bifida group reported lessparental satisfaction than parents in the able-bodied group.Mothers in the spina bifida group reported less perceived parentalcompetence, more social isolation, and less adaptability tochange; fathers in the spina bifida group reported more psychologicalsymptoms. No differences between the spina bifida and able-bodiedgroups were found with respect to marital satisfaction. Copingpredictors of adjustment tended to vary as a function of parentgender rather than group status.     

Attention and executive functions in adolescents with spina bifida

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.     

Spinal Lesion Level, Shunt Status, Family Relationships, and Psychosocial Adjustment in Children and Adolescents with Spina Bifida Myelomeningocele

Investigated whether family functioning and child psychosocialadjustment were associated with spinal lesion level and shuntstatus in 65 children and adolescents with spina bifida myelomeningocele(age range = 8–16). Mothers of children with higher lesionlevels (i.e., thoracic level) reported more attachment to theirchildren, less family conflict, and a greater willingness togrant autonomy to their offspring. Such findings support a "marginality"interpretation of the data, insofar as the least physicallyimpaired children with spina bifida exhibited the greatest familydifficulties. Based on maternal report, children with shuntsperformed more poorly in school and exhibited lower levels ofcognitive competence than children without shunts. Findingsare discussed in relation to literatures on neuropsychologicalfunctioning and psychosocial adjustment in children with spinabifida.     

Goal-Directed Behavior and Perception of Self-Competence in Children with Spina Bifida

Compared a group of school-age children with spina bifida (n= 75) between the ages of 6 and 12 years with an age- and IQ-matchedcontrol group of normal children (n = 15). As predicted, thespina bifida children spent less time using goal-directed behaviorsand more time in simple manipulation of the toys compared tothe normal children. There were no group differences betweenthe spina bifida and normal children's perceived competencebut parents of the spina bifida children rated their childrenas having lower cognitive and physical competence. Associationswere found between goal-directed behaviors and perceived self-competencefor children in the spina bifida group but not the normal group.     

Children with physical handicaps and their mothers: the interrelation of social support, maternal adjustment, and child adjustment.

Examined the interrelation of maternal adjustment, mother-child interaction, and child adjustment in 29 families of children with spina bifida and without mental retardation and in 28 families of children without handicaps. A multivariate, ecological model proposed that adjustment of mother and child depends on the adaptiveness of maternal response to the stress of the physical handicap and on the ability of mothers to create an optimal caregiving environment through mother-child interaction. Analyses examining the relationships among maternal social support, maternal psychological adjustment, and child adjustment are reported. Social support was found to be related to higher maternal psychological adjustment and to higher child adjustment, and maternal psychological adjustment was related positively to child adjustment in both groups. No significant differences were found between groups in the examined relationships or in the levels of resources and adjustment. Results underscore strengths of families of children with spina bifida in their adaptation to the stress of the handicap.     

Constructing a prospective model of psychosocial adaptation in young adolescents with spina bifida: an application of optimal data analysis

OBJECTIVE: To examine how individual- and family-level predictors in late childhood and preadolescence relate to psychosocial adaptation (i.e., scholastic success, social acceptance, and positive self-worth) in early adolescence. METHOD: This prospective longitudinal study includes 68 families of children with spina bifida and 68 comparison families of healthy children. Multimethod, multiinformant data were evaluated via optimal data analysis (ODA) and classification tree analysis (CTA) techniques. RESULTS: Factors best predicting psychosocial adaptation in early adolescence included (a) intrinsic motivation, (b) estimated verbal IQ, (c) behavioral conduct, (d) coping style, and (e) physical appearance. There were no significant group (spina bifida vs. able-bodied) effects. CONCLUSIONS: The final classification model correctly classified 77.8% of the total sample, indicating that this model had significant predictive capabilities. Results suggested that processes leading to psychosocial adaptation may be similar for youth with and without chronic illness.     

Perceived Problem-Solving Ability, Stress, and Coping in Mothers of Children with physical disabilities: Potential cognitive influences on adjustment

Examined the contribution of perceived problem-solving ability to the adjustment of mothers of children with a physical disability, in conjunction with appraised disability-related stress and approach and avoidance coping, as a further evaluation of Wallander and Varni's Disability-Stress-Coping model emphasizing cognitive processes. One hundred sixteen mothers of children, ages 2 to 20, with spina bifida or cerebral palsy completed measures of relevant constructs. Results show that the mother's appraisal of disability-related stress is strongly associated with her reported maladjustment. However, perceptions of competence in problem solving are associated with better overall adjustment. A portion of this relation appears mediated by coping styLe, as confidence in one's problem-solving ability increases so does the likelihood of selecting more adaptive coping strategies when faced with disability-related stress. Overall, consideration of perceived problem-solving ability appears to make a valuable contribution to the Disability-Stress-Coping model and to current understanding of the experience of coping with raising a child with a disability. Perceived problem-solving ability can also be linked to clinical intervention, as discussed.     

Child and Maternal Temperament Characteristics,Goodness of Fit,and Adjustment in Physically Handicapped Children

We investigated temperament characteristics and their goodness of fit as potentially important moderators of adjustment in children with congenital physical disabilities. The mothers of fifty 6-to 11-year-olds with spina bifida or cerebral palsy completed questionnaires designed to separately assess their children's and their own temperament along five dimensions (activity, distractibility, adaptability/approach, rhythmicity, and reactivity) and their children's adjustment in three areas (internalizing behavior problems, externalizing behavior problems, and social competence). We found disabled children's adjustment to be significantly worse than that of a normative comparison group. We also found significant multivariate relationships between child temperament (but not maternal temperament) and child adjustment. However, knowledge of maternal rhythmicity enhanced predictions of some child adjustment dimensions beyond that afforded by salient child temperament dimensions. The goodness-of-fit model, in which temperament characteristics of mother and child are purported to interact to influence adjustment, was not supported. Consequently, a simpler additive model, in which both child and maternal temperament are considered, appeared most useful in explaining child adjustment.     

Mental Rotation Performance in Children With Hydrocephalus Both With and Without Spina Bifida

The mental rotation ability in children with hydrocephalus and those with both hydrocephalus and spina bifida in comparison to healthy controls was investigated in this study. All groups performed a chronometric mental rotation test. Compared to children with hydrocephalus, children with both spina bifida and hydrocephalus showed an impaired mental rotation performance, demonstrated by slower reaction times. No significant performance difference was found between children with spina bifida and healthy controls. Error rates were comparable between groups indicating that the impaired mental rotation performance in children with both spina bifida and hydrocephalus is primarily due to motor impairment.     

Perceived Role Restriction and Adjustment of Mothers of Children with Chronic Physical Disability

Examined the relationship between maternal perceived role restriction,the extent to which a mother feels unable to pursue her owninterests due to responsibilities with raising a child witha chronic physical condition, and psychosocial adjustment in50 mothers with children 6–11 years old who had a chronicphysical disability. Perceived role restriction accounted fora significant increment in the variation in adjustment beyondthat contributed by objective indices of the child's disability.Neither these objective parameters of the child's disabilitynor his or her level of problem behaviors predicted perceptionsof role restriction. However, perception of social support did.The potential role of intrapersonal processes was discussedas a neglected component of a multidimensional model of adjustmentin mothers of children with a chronic physical condition.     

Observed and perceived dyadic and systemic functioning in families of preadolescents with spina bifida

OBJECTIVE: To examine dyadic and systemic family functioning across several domains (conflict, cohesion, and stress) in families of preadolescents with spina bifida in comparison to families of able-bodied preadolescents (8- and 9-year olds; n = 68 in each sample). METHODS: Mother-, father-, and child-reported questionnaire data and observational ratings of family behavior were employed. RESULTS: Findings revealed significant group and socioeconomic status (SES) differences, particularly for the observational family data. Compared to families of able-bodied children, families in the spina bifida sample were less cohesive and children from this sample were more passive during family interaction tasks. Additional analyses suggested that some of these significant associations between group status and family functioning were mediated by verbal IQ, indicating that a significant portion (42%-55%) of the overall group effect was due to variations in child cognitive functioning. Lower SES families demonstrated higher levels of observed mother-child conflict, less observed and perceived family cohesion, and more life events. Lower SES families from the spina bifida sample appear to be particularly at risk for lower levels of family cohesion. CONCLUSIONS: Findings for the spina bifida sample support a resilience-disruption view (Costigan, Floyd, Harter, & McClintock, 1997) of systemic functioning in families of children with pediatric conditions.     

Variation and expression of dihydrofolate reductase (DHFR) in relation to spina bifida

The dihydrofolate reductase (DHFR) enzyme is important for folate availability, folate turnover and DNA synthesis. The 19-bp deletion in intron-1 of DHFR has been associated with the risk of having spina bifida affected offspring, supposedly by changing DHFR gene expression. A 9-bp repeat in exon 1 of the mutS homolog 3 (MSH3) gene was recently demonstrated to be also located in the 5'UTR of DHFR and may possibly affect DHFR gene expression as well. We examined the association between these DHFR variants and spina bifida risk and investigated their effect on DHFR expression. Our study population, consisting of 121 mothers of a spina bifida affected child, 109 spina bifida patients, 292 control women and 234 pediatric controls was screened for the DHFR 19-bp deletion and the DHFR 9-bp repeat. DHFR gene expression was measured in 66 spina bifida patients, using real-time PCR analysis. In this study population, the DHFR 19-bp del/del genotype was not associated with spina bifida risk in mothers and children (OR: 0.8; 95%CI: 0.4-1.5 and OR: 1.2; 95%CI: 0.6-2.2, respectively) and both the WT/del and the del/del genotype did not affect DHFR expression relative to the WT/WT genotype (relative expression=0.89, p=0.46 and relative expression=1.26, p=0.24, respectively). The DHFR 9-bp repeat was not associated with spina bifida risk in mothers and children. DHFR expression of the 6/6 allele was 73% increased compared to the 3/3 allele, although not significantly (relative expression=1.73, p=0.09). We did not find evidence for an effect of the DHFR 19-bp deletion or 9-bp repeat on spina bifida risk in mothers and children. An effect of the 6/6 repeat genotype on DHFR expression cannot be ruled out.     

Sib risk of neural tube defect: is prenatal diagnosis indicated in pregnancies following the birth of a hydrocephalic child?

Recurrence frequencies of central nervous system malformations in sibs of probands with anencephalus or spina bifida range between 1% and 7%. The frequency of hydrocephalus among sibs of such probands is low (0.21%) but, nevertheless, is increased 2 to 5-fold when compared to general population frequencies. Anencephalus and spina bifida cystica were observed in 1.65% of sibs of children with hydrocephalus, a 2- to 8-fold increased over the population frequencies. These data indicate that some aetiological factors may be common to all three malformations. The risk figure of 1.65% for anencephalus and spina bifida in sibs born after the birth of a hydrocephalic proband constitutes sufficient indication for prenatal diagnosis by alphafetoprotein determination of the amniotic fluid.     

Latex sensitization in children with spina bifida: follow-up comparative study after two years.

BACKGROUND: Previous findings suggest that sensitization to latex in children with spina bifida is a dynamic process. OBJECTIVE: To study if changes appear in the sensitization status after withdrawal of latex. METHODS: We studied a consecutive sample of 68 children with spina bifida, by means of latex skin prick tests and quantification of serum latex-specific IgE on two separate occasions two years apart. RESULTS: Forty-four (65%) were classified as nonsensitized, 6 (9%) showed indeterminate results, and 18 (26%) were sensitized to latex, six of whom had clinical reactions to latex. They were instructed to avoid latex. In a second evaluation, 2 years later, 38 (56%) were classified as nonsensitized, 3 (4%) as indeterminate, and 27 (40%) as sensitized to latex, 11 of whom had presented latex symptoms. This meant 22% of spina bifida children demonstrated progressive sensitization, in spite of having adopted a latex-free environment at our hospital. It illustrates the progressive character of latex sensitization in these patients. CONCLUSION: Latex avoidance measures both in the medical and home settings must be stressed. We recommend that children with spina bifida should be periodically evaluated regarding latex sensitization.     

Behavioral Adjustment of Children With Hydrocephalus: Relationships With Etiology, Neurological, and Family Status

Examined the relationship of hydrocephalus and behavioral adjustmentin three groups of 5- to 7-year-old children (N = 84) with ahistory of early hydrocephalus (spina bifida, prematurity, aqueductalstenosis) and three non-hydrocephalic comparison groups (spinabifida, prematurity, normals). Results revealed no significantgroup differences on measures of behavioral adjustment and avariety of family and sociodemographic variables. Children withhydrocephalus were more likely to meet criteria for behaviorproblems, obtained lower scores on measures of adaptive behavior,and perceived themselves as less physits treatment, gender,family variables and motor skills were related to the presenceof behavior problems.     

Brief report: testing the factorial invariance of the CBCL Somatic Complaints scale as a measure of internalizing symptoms for children with and without chronic illness

OBJECTIVE: To examine the factorial invariance of the Somatic Complaints subscale of the Child Behavior Checklist as a measure of Internalizing Behavior Problems across a sample of children with and without spina bifida. METHODS: Multisample confirmatory factor analysis was used to compare mother and father report on the Somatic Complaints subscale across a sample of children with spina bifida and a matched comparison sample of able-bodied children ages 8 through 11 years (N = 68 for mother report in each group; N = 54 for father report in the spina bifida group and 53 for the able-bodied group). RESULTS: Although there were no significant between-group differences in the magnitude of factor loadings, significantly more variance in scores on the Somatic Complaints scale was unrelated to Internalizing Behavior Problems for the spina bifida group, compared to the able-bodied group. There were no between-group differences when father data were analyzed, but the latent variable of Internalizing Behavior Problems explained little variance in the Somatic Complaints scale for either group. CONCLUSIONS: Maternal report of Somatic Complaints on the CBCL does not appear to measure Internalizing Behavior Problems in the same manner across groups of children with and without spina bifida. This suggests that the Somatic Complaints subscale should be interpreted with caution when measuring Internalizing Behavior Problems within this population.