This article focuses on how the Life Course Model Web site can help family members build on the strengths of individuals with spina bifida and address areas of difficulty. A developmental perspective is adopted, which maintains that the Life Course Model Web site is useful at all stages of development, with the information provided for families at one stage of development building on the information provided for those at earlier stages of development. A brief overview is provided of relevant theories that supported the development of the Life Course Model. There is a review of the literature on the adjustment of families of individuals with spina bifida and the psychosocial adjustment of affected youth. How families may benefit from engagement with the 3 content areas covered by the Web site is discussed, namely child health and the transfer of medical management from parent to child (health/self-management), the development of social relationships (social relationships), and the achievement of milestones during emerging adulthood, including achievements in the areas of education and employment (education, employment, and income support).
Spina bifida (SB) is a common congenital birth defect that has a significant multisystemic effect on the physical, neurocognitive, psychological, and social functioning of affected individuals. It is also well known that the clinical symptoms of SB place considerable physical, psychological, and social demands on the family members involved. Despite the pervasive effect that this condition has on family members and affected individuals, there are few family-based interventions for this at-risk population. A recent review of the literature on family interventions failed to identify any evidence-based interventions that focused on improving the psychosocial functioning of families of youth with SB. Thus, the Web-based product that is the focus of this special issue is a welcome offering for families who currently lack such a resource.
Although Web-based products for families of youth with SB have not been available until now, there are family-oriented products for populations other than SB, including chronic pain, traumatic brain injury, cancer, and encopresis. Research that examines the usefulness of such sites has revealed that parents and affected youth find these Web resources to be useful as they work on personal goals and that their knowledge of their condition increases with increased use. However, in some cases use has been lower than expected, perhaps because recruitment for the studies was timed inappropriately (ie, too close to diagnosis) or because the modules were too time intensive. Family use is higher if hands-on training is provided.
This article focuses on how this new Web site, to be housed at the Spina Bifida Association of America (SBA) and based on the Life Course Model discussed throughout this issue, can help family members build on the strengths of individuals with SB and address areas of difficulty. Moreover, and consistent with the developmental orientation of the Life Course Model, we adopt a developmental perspective in this paper. That is, we maintain that the Life Course Model Web site is useful at all stages of development, with the information provided for families at one stage of development building on the information provided for those at earlier stages of development.
First, a brief overview is provided of relevant theories that supported the development of the Life Course Model, on which the Web site is based. Next, the literature on the adjustment of families of individuals with SB is reviewed as well as the literature on the psychosocial adjustment of affected youth. Specifically, an overview is provided of areas of difficulty for which families are most likely to seek help when using this product. We also expect that families will use the product to build on areas of existing strength. How families may benefit from engagement with the 3 content areas covered by this Web site is also discussed, namely: (1) child health and the transfer of medical management from parent to child (health/self-management), (2) the development of social relationships (social relationships), and (3) the achievement of milestones during emerging adulthood, including achievements in the areas of education and employment (education, employment, and income support).
Incorporation of family-centered care and the chronic care principles into the Life Course Model
The article by Swanson in this issue provides details on the conceptual thinking that incorporated principles of normal child development and the international classification of functioning, health and disability (ICF) into a framework, termed the Life Course Model. Concisely, the Life Course Model assumes: (1) for optimal development in different domains, children need to attain a sequence of milestones, which are common to all children; (2) the ICF contributes the functional domains of importance and the principle that function is an outcome of the interaction between underlying impairment (eg, cognitive deficit, leg weakness) and the surrounding environment (family, school, community).
The Life Course Model is also anchored in 2 interlocking models of health care delivery, family-centered care (FCC) and the chronic care model (CCM), both of which underscore the central importance of partnering with families to enhance medical and psychosocial outcomes for children with special health care needs, including those with SB. FCC has deep roots in the pediatric literature, particularly in research and clinical care for children with special health care needs and their families. The key elements of FCC include (1) encouraging parental involvement in decision-making; (2) fostering empowerment and self-management capacity in families; (3) attending to the needs of all members of the family unit; and (4) providing culturally respectful services. FCC also emphasizes tailoring services to each family’s identified strengths, priorities, and needs.
This Life Course Model is consistent with the fundamental principles of FCC through its emphasis on equipping key stakeholders (including individuals with SB and their parents) with scientific data, assessment tools, and community resource information in 3 content domains: health/self-management, social relationships, and education/income support. The complementary sources of information presented on the Life Course Model Web site ideally maximize parents’ capacity for informed decisions about their child’s health care and position them to advocate for their child’s and family’s needs across the lifespan.
Establishment of the Life Course Model and construction of the Web site were similarly guided by aspects of the CCM, which suggests that productive interactions in health settings develop from “informed, activated patients and prepared, proactive practice teams.” (p3) Development of the CCM was sparked by increased recognition of the shortcomings of the health system in attending to psychosocial demands associated with chronic health conditions, namely the physical, social, and psychological stressors experienced by both the affected individual and surrounding family. To address this apparent gap in clinical care, the CCM provides evidence-based guidelines for improving health services at multiple levels of the health system (community, organization, practice, and patient levels). Specifically, the model purports that ideal patient-provider interactions, reduced health care costs, enhanced patient satisfaction, and optimal health outcomes result from proper health care organization and delivery system design, systematic use of clinical information systems, health care provider decision support, patient and caregiver self-management support, and community linkages and resources. Another aspect of CCM is the shared management model, in which professionals and families intentionally shift care responsibility across the life course, with the individual assuming full responsibility in adulthood.
The Life Course Model Web site addresses multiple dimensions of the CCM, namely (1) provider decision support, (2) patient and caregiver self-management support, and (3) community linkages and resources. Health care providers may access the Web site to learn teaching tips as they strive to partner with individuals with SB and their families on topics of interest. Content on health/self-management, social relationships, and education/income support is presented from a lifespan perspective, which likely addresses immediate as well as long-term concerns identified by parents and individuals with SB. In the following sections, the extant literature on the adjustment of family members who have a child with SB and the psychosocial adjustment of youth with SB is reviewed, highlighting areas of difficulty that are addressed by the Life Course Model Web site. Next, how families may benefit from the Web site is outlined, including how this novel resource might maximize individual and family self-management.
Review of past research on family functioning in SB
Family relationships are particularly salient and influential social relationships for youth with SB, given that children with SB tend to be more socially isolated from their peers than are typically developing children. Further, we are interested in family relationships because SB affects not only the child but also the parents and other family members. Given the pervasive effect of this condition, we were also interested in discussing the level of psychosocial adjustment in such individuals across multiple adjustment domains (eg, internalizing symptoms, externalizing symptoms, self-concept).
Family Functioning
Holmbeck and colleagues published a review of research that examined the effect of SB on family functioning. In general, the findings of past work support a disruption-resilience view of family functioning. That is, SB seems to disrupt some aspects of family and parent functioning for many families, but such families also tend to show considerable resilience across other adjustment domains. A significant number of families in which there are children who have SB report difficulties in maintaining clear roles and responsibilities in the family system (23% in the clinically problematic range ). With respect to risk factors, Holmbeck and colleagues found that families of youth with SB who were also from lower socioeconomic status (SES) backgrounds were particularly at risk for lower levels of family cohesion, supporting a cumulative risk view of such families (ie, SB status and lower SES have additive effects on family functioning). Given this situation, it is particularly important to market this Web site to families from low-income backgrounds and to address issues related to the availability of computer resources in this subpopulation.
Families of youth with SB do not seem to change with the development of their offspring in the same manner as is found with typically developing youth. For example, with respect to family conflicts, Coakley and colleagues found that, unlike their typically developing peers, families of youth with SB did not show normative increases in family conflict as a function of pubertal development. These investigators speculated that families of youth with SB may be less responsive to developmental change. In support of this attenuated response to development hypothesis, Jandasek and colleagues conducted longitudinal growth analyses in the age period of 9 to 15 years and found that family conflict intensity increased during this early adolescent age range in families of typically developing youth but not in families of children with SB. Further, parents of youth with SB are less likely to discuss issues of sexuality with their offspring than are parents of typically developing youth. Therefore, this Web site aims to serve as a resource to help parents be more responsive to maturational changes in their child with SB.
Adjustment of Parents and Parenting Behaviors
Despite the low levels of family dysfunction at the family systems level, it seems that a sizable minority of parents of children with SB exhibit clinical levels of global psychological distress (eg, anxiety, depressive symptoms, somatic complaints ). Although most studies that report on parental functioning have focused on maternal functioning, one study of fathers of children with SB indicated that fathers exhibited higher levels of global distress compared with fathers from comparison families. In a recent meta-analysis of 15 studies, Vermaes and colleagues found medium to large effect sizes for the effect of SB on mother and father’s psychological adjustment, with larger effects sizes for mothers ( d = .73) than for fathers ( d = .54).
Across several studies, parents of children with SB tend to experience more stress in their roles as parents than do comparison parents. Typically, such parents feel less satisfied and competent as parents, feel more isolated, are less adaptable to change, and hold less optimistic views about the future than comparison parents. Parents who are single, socially isolated, older, or from an ethnic minority or a low SES background are particularly at risk for such outcomes.
With respect to parenting behaviors, it has been found that increases in parental responsiveness are associated with increases in adaptive coping strategies in youth with SB (eg, problem-focused coping ). However, parents of children with SB tend to exhibit higher levels of intrusiveness, psychological control, and authoritarian parenting (ie, parenting that undermines the autonomy development of their offspring ) and these behaviors tend to be linked with less desirable child outcomes. Specifically, higher levels of intrusiveness (sometimes referred to as overprotectiveness) tend to be associated with lower levels of decision-making autonomy, which are in turn related to higher levels of psychosocial difficulties. Thus, parents may find this product useful in learning how to avoid certain forms of parenting that may not be beneficial to their child, which seem to be more common in families of children with SB.
Adjustment of Siblings
Few studies have examined the functioning of siblings of children with SB. Findings have been contradictory: an early study using teacher reporting found a 4-fold greater likelihood of adjustment problems for siblings relative to a comparison sample, whereas a later study of siblings of youth with SB reported no differences in self-concept compared with siblings of typically developing youth. Qualitative research has identified both positive and negative outcomes related to having a sibling with SB. For example, siblings report significant levels of concern for the health of their sibling with SB, emotional upset in relation to their sibling’s experience with discrimination, teasing, and bullying, and sadness related to the lack of opportunities to engage in physical activities with their sibling with SB. Siblings have also identified some positive effects, such as increased empathy for their sibling and a greater appreciation for their own physical abilities.
The behavioral and psychological functioning of siblings has been found to be significantly associated with SES, family cohesion, perceptions of social support, and their knowledge of and attitudes toward the illness. For siblings of children with SB, more positive attitudes toward SB, greater family satisfaction, lower levels of sibling conflict, and increased social support from classmates significantly predicted higher levels of self-concept and prosocial behavior, and lower rates of behavior problems. In this study by Bellin and colleagues, family satisfaction was the only significant predictor across all 3 sibling adjustment outcome measures, suggesting that family variables may be particularly salient for sibling adjustment. Therefore, parents of youth with SB may also use this Web site tool to learn of ways to protect their typically developing offspring from psychosocial difficulties.
Review of past research on family functioning in SB
Family relationships are particularly salient and influential social relationships for youth with SB, given that children with SB tend to be more socially isolated from their peers than are typically developing children. Further, we are interested in family relationships because SB affects not only the child but also the parents and other family members. Given the pervasive effect of this condition, we were also interested in discussing the level of psychosocial adjustment in such individuals across multiple adjustment domains (eg, internalizing symptoms, externalizing symptoms, self-concept).
Family Functioning
Holmbeck and colleagues published a review of research that examined the effect of SB on family functioning. In general, the findings of past work support a disruption-resilience view of family functioning. That is, SB seems to disrupt some aspects of family and parent functioning for many families, but such families also tend to show considerable resilience across other adjustment domains. A significant number of families in which there are children who have SB report difficulties in maintaining clear roles and responsibilities in the family system (23% in the clinically problematic range ). With respect to risk factors, Holmbeck and colleagues found that families of youth with SB who were also from lower socioeconomic status (SES) backgrounds were particularly at risk for lower levels of family cohesion, supporting a cumulative risk view of such families (ie, SB status and lower SES have additive effects on family functioning). Given this situation, it is particularly important to market this Web site to families from low-income backgrounds and to address issues related to the availability of computer resources in this subpopulation.
Families of youth with SB do not seem to change with the development of their offspring in the same manner as is found with typically developing youth. For example, with respect to family conflicts, Coakley and colleagues found that, unlike their typically developing peers, families of youth with SB did not show normative increases in family conflict as a function of pubertal development. These investigators speculated that families of youth with SB may be less responsive to developmental change. In support of this attenuated response to development hypothesis, Jandasek and colleagues conducted longitudinal growth analyses in the age period of 9 to 15 years and found that family conflict intensity increased during this early adolescent age range in families of typically developing youth but not in families of children with SB. Further, parents of youth with SB are less likely to discuss issues of sexuality with their offspring than are parents of typically developing youth. Therefore, this Web site aims to serve as a resource to help parents be more responsive to maturational changes in their child with SB.
Adjustment of Parents and Parenting Behaviors
Despite the low levels of family dysfunction at the family systems level, it seems that a sizable minority of parents of children with SB exhibit clinical levels of global psychological distress (eg, anxiety, depressive symptoms, somatic complaints ). Although most studies that report on parental functioning have focused on maternal functioning, one study of fathers of children with SB indicated that fathers exhibited higher levels of global distress compared with fathers from comparison families. In a recent meta-analysis of 15 studies, Vermaes and colleagues found medium to large effect sizes for the effect of SB on mother and father’s psychological adjustment, with larger effects sizes for mothers ( d = .73) than for fathers ( d = .54).
Across several studies, parents of children with SB tend to experience more stress in their roles as parents than do comparison parents. Typically, such parents feel less satisfied and competent as parents, feel more isolated, are less adaptable to change, and hold less optimistic views about the future than comparison parents. Parents who are single, socially isolated, older, or from an ethnic minority or a low SES background are particularly at risk for such outcomes.
With respect to parenting behaviors, it has been found that increases in parental responsiveness are associated with increases in adaptive coping strategies in youth with SB (eg, problem-focused coping ). However, parents of children with SB tend to exhibit higher levels of intrusiveness, psychological control, and authoritarian parenting (ie, parenting that undermines the autonomy development of their offspring ) and these behaviors tend to be linked with less desirable child outcomes. Specifically, higher levels of intrusiveness (sometimes referred to as overprotectiveness) tend to be associated with lower levels of decision-making autonomy, which are in turn related to higher levels of psychosocial difficulties. Thus, parents may find this product useful in learning how to avoid certain forms of parenting that may not be beneficial to their child, which seem to be more common in families of children with SB.
Adjustment of Siblings
Few studies have examined the functioning of siblings of children with SB. Findings have been contradictory: an early study using teacher reporting found a 4-fold greater likelihood of adjustment problems for siblings relative to a comparison sample, whereas a later study of siblings of youth with SB reported no differences in self-concept compared with siblings of typically developing youth. Qualitative research has identified both positive and negative outcomes related to having a sibling with SB. For example, siblings report significant levels of concern for the health of their sibling with SB, emotional upset in relation to their sibling’s experience with discrimination, teasing, and bullying, and sadness related to the lack of opportunities to engage in physical activities with their sibling with SB. Siblings have also identified some positive effects, such as increased empathy for their sibling and a greater appreciation for their own physical abilities.
The behavioral and psychological functioning of siblings has been found to be significantly associated with SES, family cohesion, perceptions of social support, and their knowledge of and attitudes toward the illness. For siblings of children with SB, more positive attitudes toward SB, greater family satisfaction, lower levels of sibling conflict, and increased social support from classmates significantly predicted higher levels of self-concept and prosocial behavior, and lower rates of behavior problems. In this study by Bellin and colleagues, family satisfaction was the only significant predictor across all 3 sibling adjustment outcome measures, suggesting that family variables may be particularly salient for sibling adjustment. Therefore, parents of youth with SB may also use this Web site tool to learn of ways to protect their typically developing offspring from psychosocial difficulties.
Review of past research on psychosocial functioning in SB
Research on Children and Adolescents
Previous studies have shown that youth with SB are at risk for exhibiting higher levels of internalizing symptoms (eg, depression, anxiety) and lower levels of self-concept than comparison children. Those with hydrocephalus often exhibit difficulties in certain areas of cognitive functioning and school performance (eg, arithmetical, nonverbal cognitive skills ;). Such children are also more likely to exhibit attention and concentration difficulties in school settings and tend to score at the low end of the average range of intelligence.
To date, more work has been done in evaluating children with SB in the areas just noted than has been conducted in the area of social adjustment. This lack of attention is surprising given that this area of psychosocial functioning is problematic for most children with SB. Youth with SB, compared with typically developing youth and those with other chronic conditions, tend to be socially immature and passive, to have fewer friends, to be less likely to have social contacts outside school, and to date less during adolescence, and these difficulties seem to be maintained over time.
The degree to which an adolescent exhibits decision-making autonomy in both medical and nonmedical areas is another highly salient developmentally oriented variable for these youth and their families. Typically developing adolescents view more issues as falling within their own decision-making jurisdiction than they did during childhood and they are also increasingly likely to question the legitimacy of parental authority. Holmbeck and colleagues’ findings on youth with SB run contrary to this typical developmental trend. Specifically, findings revealed that children and adolescents with SB (and especially boys and those with lower levels of intelligence) tend to be more dependent on adults for guidance, less likely to exhibit behavioral autonomy at home, less likely to exhibit intrinsic motivation at school, and less likely to express their own viewpoints during observed family interactions. Variation in intrinsic motivation (ie, interest in learning and mastery, curiosity, preference for challenge) proved to be the most robust predictor of psychosocial adaptation (ie, scholastic success, social acceptance, and positive self-worth) in a study by Coakley and colleagues.
Research on Emerging Adults
Emerging adulthood is a critical period in the life of older adolescents with SB (First World Congress on SB Research and Care, March 2009, Orlando, FL, USA ). In general, many young adults with SB are capable of high levels of independent functioning across multiple domains but most have not been successful in fully engaging in the larger community of typically developing emerging adults. In this section, we review findings related to many of the major milestones of emerging adulthood (ie, psychosocial adjustment, educational achievement, and employment and vocational outcomes).
Regarding psychosocial adjustment, emerging adults with SB, like their younger counterparts, are at risk for depressive symptoms and anxiety. Regarding educational outcomes, emerging adults with SB are less likely to go to college (41%–49% of individuals with SB go to college vs 66% of typically developing youth ). With respect to vocational outcomes, recent studies report rates of full- or part-time employment ranging from 36% to 41% (MH Bellin and colleagues. Gender differences in self-management, community integration, and quality of life in transition-age individuals with spina bifida, unpublished manuscript, 2010), which are significantly lower than those found in typically developing youth (ie, roughly 75% ) and in those with other chronic conditions (eg, asthma, cancer; 68% to 78% ).
Little is known about factors that predict whether or not an emerging adult with SB is able to go to college and become employed. Studies that have been conducted on individuals with SB have tended to focus only on demographic or medical severity predictors. For example, Liptak and colleagues found that communication problems, difficulties with managing responsibilities, lower levels of parental education, and higher rates of parental unemployment were associated with poorer social, vocational, and educational transitions. Bellin and colleagues (Gender differences in self-management, community integration, and quality of life in transition-age individuals with spina bifida, unpublished manuscript, 2010) found that young adult men with SB were more likely to work than women, but that women were more likely to live independently. With respect to medical severity, Hetherington and colleagues found that spinal lesion level and number of shunt revisions were related to employment outcome (with higher lesion levels and more shunt revisions being related to worse occupational outcome; Barf and colleagues found similar results in the Netherlands.
In the absence of data, many have speculated about why young adults with SB are less likely to be successful in negotiating these emerging adulthood milestones. For example, some have described the complexities in managing real world responsibilities with a chronic physical condition, including transportation difficulties and issues related to accessibility. Other explanations for these developmental delays focus on financial concerns (including lack of health insurance ), lack of job training and vocational rehabilitation services, restricted experiences with self-management, employment discrimination, stigmas related to physical appearance, and a lack of autonomy-related socialization in early childhood. Many of these issues are addressed with the Life Course Model Web site resource and such content is likely useful for families.
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