Social Functioning in Children With Spina Bifida: An Integrative Review

Abstract

Background:

Spina bifida (SB) is a congenital disorder often leading to disability and limited social participation. Children with SB face challenges engaging at home, school, and in the community. This review examined the types and degrees of social functioning in children with SB.

Methods:

Researchers searched 5 databases (CINAHL, MEDLINE, PubMed, EMBASE, and PsycINFO) for studies of various designs, both qualitative and quantitative, published between 2000 and 2025 that involved children with spina bifida (SB) aged 18 years or younger.

Results:

Findings revealed restricted participation with peers and family due to continence issues, motor impairments, stigma, and cognitive difficulties. Four domains of social functioning emerged: school and community participation, peer relationships and freindships, family interactions and parental role, and independence and autonomy.

Conclusions:

Children with SB demonstrate reduced social functioning. More research is needed to clarify the impact of parental expectations on social outcomes.

Introduction

Spina bifida (SB) is the second most common birth defect globally, behind congenital heart abnormalities. ¹ It is a congenital neural tube defect characterized by incompletely closed spinal column due to incomplete closure of the neural tube. ² The severity of SB and the intensity of the associated physical challenges are determined by the size and location of the opening in the spine and the damage to the underlying nerves. A range of physical symptoms, including loss of mobility, incontinence, and hydrocephalus, can be observed in children with SB. ³ Physical impairment invariably affects other dimensions of the lives of children with SB, such as their psychological, social, and intellectual capacity. The challenges that children with SB face have long-term implications. ⁴ Social functioning in children with spina bifida refers to their capacity to form and maintain age-appropriate peer relationships and engage adaptively in social environments, an ability shaped by underlying social cognitive skills (eg, joint attention and perspective-taking), executive functions (eg, attention and planning), and behavioral autonomy. ⁵ However, these children often face challenges: they may display social immaturity and passivity, have fewer interactions outside school, rely more on adult support, and struggle with the neurocognitive processes needed to adjust socially. The consequences are particularly devastating for families of children with SB and, subsequently, for communities. Longitudinal, multimethod evidence by Holbein et al indicates that neuropsychological factors, particularly attention and executive function, are the strongest predictors of later social skills in youth with spina bifida, often explaining more variance than family or health measures. ⁵ Moreover, SB poses a substantial burden on healthcare systems. Regarding healthcare expenses, a 2013 study of a nationwide inpatient sample including all ages in the United States revealed that hospital stays linked to birth abnormalities of the brain and spine, including SB, cost more than $1.6 billion. ⁶ Grosse et al estimated that a person with SB in the United States will pay $791 900 for lifetime care, comprising $513 500 in medical costs, $63 500 in developmental services and special education costs, and $214 900 in caregiver time costs. ⁷

Children and teenagers with chronic illnesses often have decreased social functioning compared to those without chronic illnesses. ⁸ Many chronic diseases restrict the type and degree of physical exercise, social interaction, and school attendance; therefore, this restriction may be caused by many elements. These limitations may be attributable to physical restrictions, prolonged or repeated hospitalizations, frequent doctor’s appointments, or special transportation requirements. Low levels of community and preschool/school engagement were observed among children with spina bifida. Low levels of participation, dependency in day-to-day living, and delayed social development are frequently noted effects of spina bifida. Due to advances in neuropsychological research, we now know more about the cognitive consequences of spina bifida on language, perception, attention, memory, and executive functioning than we did 30 years ago. ⁹

Many children and adolescents with SB have poor social adjustment throughout development stages. ¹⁰ Previous research has focused on the limitations and challenges children with SB face when engaging in social functioning. For example, continence issues (ability to control bowel and bladder movements) were a major limitation for most participants in the studies by Fischer et al ¹² and Kelly et al. ²³ Incontinence problems in children with SB range from minimal to significant, depending on the level of the lesion. Because they were ashamed and felt compelled to conceal it from their peers, children with SB saw their incontinence as a serious problem in addition to the physical pain they experienced. Recent research indicates that 87% of people with spina bifida have bowel incontinence, and fewer than 30% have bowel continence. Urinary continence problems afflict 48% to 76% of children with the condition. ¹¹ It can significantly hinder their social lives, limiting their participation in activities, reducing their peer relationships, and negatively impacting their self-esteem. ¹² Similarly, children with spina bifida experience a decline in their quality of life and health-related quality of life as the severity of their bowel incontinence symptoms increases.

Much of the literature has focused on medical perspectives, while children’s own voices remain underrepresented. Yet, children’s perspectives are crucial for understanding how they interpret stigma, negotiate autonomy, and construct their identities. For instance, a qualitative study of Palestinian children with SB revealed experiences of vulnerability, dependency, and exclusion, but also highlighted resilience fostered through family and religious support. Such findings underscore that social functioning is deeply shaped by cultural, economic, and political contexts. ¹³ Children in high-income countries may benefit from inclusive education and rehabilitation programs, whereas those in low-resource or conflict-affected settings face compounded barriers of poverty, stigma, and limited services. Palestinian children with SB have reported vulnerability and exclusion, but also resilience through family and community support. ¹⁴

This review aimed to assess the types and degrees of social functioning in children with spina bifida. Through clarifying the strengths and gaps in the existing literature, the review underscores key opportunities to enhance both research efforts and the psychosocial and developmental services provided to these children.

Methods

Data Sources

Whittemore and Knafl’s framework provided the basic conceptual structure for this integrative review. ¹⁵ The 5 stages: 1. problem identification, 2. literature search, 3. data evaluation, 4. data analysis, and 5. Presentation. These were rigorously followed to ensure methodological transparency and trustworthiness.

During problem identification, the research question was refined to examine the dimensions of social functioning in children with spina bifida (SB). In the literature search stage, comprehensive searches were conducted across 5 major databases (CINAHL, MEDLINE, PubMed, EMBASE, and PsycINFO) using controlled vocabulary and Boolean operators to maximize inclusiveness. The data evaluation stage employed Olsen and Baisch’s ¹⁹ quality appraisal tool to assess methodological quality. The critical appraisal was conducted independently by 2 researchers (EA [Reviewer 1] and SA [Reviewer 2]). Any disagreements were resolved through discussion and consensus. For data analysis, a constant comparative technique was applied to identify patterns and themes across studies, integrating both qualitative and quantitative findings to develop synthesized domains. Finally, the presentation stage followed PRISMA guidelines to ensure clear reporting of the search and selection process.

To enhance rigor and confirmability, several strategies were employed. Transparent reporting was maintained through detailed documentation of database searches, inclusion/exclusion criteria, and quality assessment procedures. Reflexivity was observed by acknowledging potential author biases during data extraction and synthesis, with regular discussions to ensure that interpretations were grounded in evidence rather than assumptions. Triangulation of data sources including qualitative, quantitative, and mixed-method studies strengthened the comprehensiveness and depth of interpretation. In addition, peer debriefing among team members was conducted throughout the synthesis process to verify emerging themes and interpretations. These steps collectively enhanced the credibility, dependability, and confirmability of the review findings.

This allowed more diverse articles in the literature to be included than a systematic review would have permitted. The databases searched for relevant articles included CINAHL, MEDLINE, PubMed, the Excerpta Medica Database (EMBASE), and PsycINFO. The information is summarized in the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) diagram (Figure 1). ¹⁶ The terms used were spina bifida, family functioning, social participation, social adjustment, and social functioning. Since this study is a review paper and did not involve the collection of new data from human participants or animals, Institutional Review Board (IRB) approval and informed consent were not required.

Figure 1.

PRISMA diagram.

Inclusion and Exclusion Criteria

The inclusion criteria were as follows: (1) research involving children with SB aged ≤ 18 years and/or their parents; (2) published in English between 2000 and 2025; and (3) addressed the participation of children with SB in social functioning at home, at school, with family, or with friends. Children with spinal cord injury (SCI) and typically developing peers were included in this review and compared with children with spina bifida. Studies were excluded if they failed to report relevant information on the social functioning of children with SB or if they focused exclusively on adults with SB. We focused on children with SB aged ≤ 18 years because the foundations for social functioning are generally developed during childhood, and adolescents tend to struggle with their social skills as they transition into adulthood. ¹⁷ Social functioning is particularly important for the development of social skills in children with SB. We limited the review of studies to between 2000 and 2025 to focus on more recent findings regarding children’s social functioning and skills.

Study Selection

In consideration of the PRISMA (2021) guidelines, the initial screening of titles and abstracts identified 216 potentially useful articles. Of these, 96 were duplicates and 104 did not meet the inclusion criteria, resulting in the exclusion of 200 articles. Sixteen articles that met the inclusion criteria were reviewed, and their data were analyzed and synthesized. Figure 1 summarizes the article selection process. ¹⁷

Study Synthesis

A literature review matrix (Table 1) was developed and used to organize and analyze the information from 16 articles. ¹⁸ Relevant data were extracted from each study, organized in ascending chronological order, and synthesized using a review matrix.

Table 1.

Literature Review Matrix of Selected Studies.

Study	Purpose of study	Design & sample	Relevant measures & description	Results	Quality appraisal
Kristén et al (2003) 20 Sweden	Describe parents’ perceptions about the influences of participation in a sports program on children and adolescents with physical disabilities	Qualitative, phenomenological study 20 parents of children with disabilities aged from 9-15 years old. The exact number of children with SB were not mentioned.	Interview with parents regarding the influences of participation in a sports program on their children and adolescents with physical disabilities.	• Three thematic categories emerged from interviews: achieving good health, being part of a social group, and learning a sporting activity. • The parents believed that these sporting activities could foster their children’s learning and social skills while promoting overall health and well-being. • Descriptive analysis revealed that parents viewed sports as health education and as a way for their children to increase their participation in society. The findings also show that the learning process was important for empowering the children to influence their life situation.	Study type = 3 Sampling = 2 Method = 0 Analysis = 2 Total score = 7
Boudos and Mukherjee (2008) 24 USA	Examine baseline participation of people with spina bifida (SB) in several domains (eg, school, employment, community activities, physical activity, and peer social relationships) and identify barriers to community participation.	Quantitative descriptive 101 (10-32 years old) 31 (10-17 years old)	Interviews with youth and young adults or parents about barriers to community participation. Chart review of medical condition, function/mobility, etc.	• Lack of family support was identified as a major obstacle to community involvement. • The majority of patients (77%) were in high school, but 34% of those who completed high school did not pursue further education. Only 19% of the patients completed a college degree. Among patients 18 years and older, 14% employed full time, while 10% are working part time. In terms of community participation, 22% participated at least once a week, while 54% were not involved in any activity. Regarding physical activity, 20% of the patients engaged in high-frequency exercise, while 58% did not participate in any physical activity at all. In terms of peer relations, 58% met with friends at least once a week, while 32% did not interact with friends outside of school or other activities	Study type = 3 Sampling = 2 Method = 1 Analysis = 3 Total score = 9
Liptak et al (2010) 30 USA	Describe outcomes and identify factors that affect social participation in youth and young adults with spina bifida.	Quantitative, population-based, prospective study 130 youth and young adults with SB (19.3 mean age)	Children’s Assessment of Participation and Enjoyment (CAPE) • Two domains : Informal and Formal • Five dimensions assessed for each domain: diversity, intensity, with whom, where, and enjoyment	• Outcomes: Young adults with SD were compared to others with cerebral palsy (CP), asthma, and learning disabilities (LD). Those with SB (39%) were less likely to (a) be competitively employed than those with LD (78%) and asthma (73%), (b) have a driver’s license (30%) than those with asthma (60%) and LD (70%), and spend time with friends (15%) than those with asthma (16%) and LD (38%). • Factors: Each outcome included factors associated with it including factors related to body function and structure (eg, use of mobility aids), activities (eg, using arms or hands), and environment (eg, receiving services/supported school).	Study type = 4 Sampling = 2 Data collection Method = 1Analysis = 3 Total score = 10
Kelly et al (2011) 23 USA	Examine how social participation of youth with SB varies by age and related factors (gender, family characteristics, and SB-related factors)	Quantitative, cross- sectional study 63 youth with SB ages 2-18 years and/or their caregivers	Children’s Assessment of Participation and Enjoyment (CAPE) • Participation of children with disabilities in 4 areas: recreation, physical, social, and skill-based	• Older youth with SB participated less in recreational, physical, and skill-based activities than younger youth with SB. • Participation did not differ by child gender, caregiver marital status, or caregiver education. However, youth whose caregivers were employed participated more often n social activities. • Participation did not differ by child’s motor level, ambulation, or bladder and bowel issues. However, youth who did not have a shunt participated more often in physical and skill-based activities.	Study type = 4 Sampling = 1 Data collection Method = 1Analysis = 3 Total score = 9
Devine et al (2012) 27  USA	Characterize dyadic and general friendships of youth with SB	Quantitative, longitudinal study 106 youth with SB (8-15 years old) and 106 peers (6-17 years old)	• General Friendship Interview (social adjustment, social performance, and social skills.) • Dyadic Friendship Interview (social adjustment, social performance, and social skills with in specific didactic friendship) Questionnaires: • Friendship Qualities Scale (companionship, conflict, help, security, and closeness) • Children’s Self Efficay for Peer Interaction Scale (CSPI) (perceived self-efficacy in social situations) • Emotional Support Questionnaire (ESQ) (quality of friendship in general)	• Many similarities were observed between youth with SB and their peers. More youth with SB considered peers to be their best friends than peers considered youth with SB to be their best friends. Also 9.4% of youth showed to have dyadic relation with their teacher. • Youth with SB spent fewer days with friends and showed lower levels of security, companionship, and emotional support from friends and families when compared to chosen peers without SB. • Youth with SB reported that they perceive low level of security in their friendships as compared to their non-SB peers. • Youth with SB and peers reported engaging in certain basic skills, such as asking friends to get together and choosing an activity, at similar rates. However, peers reported greater social self-efficacy. Additionally, youth with SB reported engaging in a negative social behavior (ie, teasing or being mean to other children) more frequently than selected peers. • Youth also reported that they received a low degree of emotional support from their peers.	Study type = 4 Sampling = 3 Data collection Method = 1Analysis = 3 Total score = 11
Flanagan et al (2013) 32 USA	Describe psychosocial outcomes of children and adolescents with early-onset spinal cord injury (SCI) or SB	Quantitative, cross-sectional study 137 youth including 54 youth with SB and 83 youth with SCI (aged 5-18 years old)	Availability and Participation Scale School Function Assessment (SFA) • Two scales: availability (27 items) and participation (29 items) can you describe dimensions/areas? • Three parts: participation, task support, and activity performance	• Children with SCI and SB showed similar levels of participation intensity, with scores of 2.1 (of a possible 7) for children with SB and 2.2 for children with SCI. • Children with SB participated in fewer activities than those with paraplegic SCI.	Study type = 4 Sampling = 1 Data collection Method = 1Analysis = 3 Total score = 9
Peny-Dahlstand et al (2013) 21 Sweden	Evaluate how children with SB participate in school-related activities and explore whether their motor and process skills for task performance were related to their level of active school participation	Quantitative, cross-sectional study 50 children with SB (aged 6-14 years old) and their teachers	• Availability and Participation Scale (adapted Swedish version) to rate the child’s frequency of participation in different activities during the school day (respondent’s perception of the degree of availability of the activity and the degree of the child’s participation in the same activity)	• Among children with SB, 89.6% had a low level of active participation and involvement in school activities even though their frequency of participation was high. • Children discuss their problems with friends but more difficult to discuss bladder and urinary issues. • Motor and process skills were found to be the most significant determinants of participation. • The teachers in the study reported that most children (89.6%) were restricted in active participation in school related activities.	Study type = 3 Sampling = 2 Data collection Method = 1Analysis = 2 Total score = 8
Essner et al (2014) 22  USA	Test a model of social competence in youth with SB in which debilitating condition factors (eg, greater pain intensity, weight status, and motor dysfunction) would be associated with reduced social activity involvement, which would, in turn, be linked to lower levels of social competence in youth with SB.	Quantitative, longitudinal study 108 youth with SB (aged 8-15 years old) and their parents and teachers	• Interviews • Pain intensity—Visual Analog Scale (VAS) • Gross Motor Function Classification System for SB (youth’s degree of self-initiated movement) • Weight—BMI percentile • Activities Competence Scale of the Child Behavior Checklist (CBCL) (level of participation in social activities) • Social Acceptance subscale of the Self-Perception Profile for Children (SPPC)—youths’ perception of their own social acceptance by peers as well as parents and teachers’ perceptions.	• Association between greater pain and lower social competence was partially explained by decreased social activity involvement • Greater pain intensity reduced social activity involvement • Association between social competence and pain decreased with greater social activity involvement. • Decreased social activity showed a significant negative effect on social competence.	Type of study = 4 Sampling = 3 Method = 1 Analysis = 3 Total score = 11
Bloemen et al (2015) 31 Netherlands	Describe both personal and environmental factors important for participation in physical activity as experienced by children with SB and their parents, with the goal of developing better intervention strategies for improving participation in physical activity among youth with SB	Qualitative, study 13 parents of children with SB (aged 4-7 years old) 31 parents of children with SB (aged 8-18 years old) and their parents (n = 31)	Semi structured interviews and focus groups Themes constructed using the Physical Activity for Persons with a Disability (PAD model)	• Personal factors related to physical activity participation in children with SB included intention to be physically active, attitude toward physical activity, health conditions such as bowel and bladder care, and facilitators and barriers within the child such as competence in skills and sufficient fitness. • Environmental factors related to children’s physical activity participation included social influence such parents’ beliefs about the importance of physical activity. As with personal factors, there were also factors and barriers within the environment such as support from others, use of assistive devices for mobility and care, having information related to possibilities for adapted sports, and access to sports facilities.	Study type = 3 Sampling = 3 Data collection Method = 1Analysis = 3 Total score = 10
Fischer et al (2015) 12 Canada	Explore the experiences of children and young people with spina bifida (SB) around continence issues, social participation and peer relationships, in order to identify potential areas of support healthcare professionals can provide.	Qualitative, phenomenological study 11 children with SB (aged 6-18 years old) 10 parents	• Interviews • Children asked about the effects of SB on their activities with friends, need for assistance to go to the bathroom, & wearing assistive devices to stay dry. Parents asked about restrictions related to SB, and reliance on parents or major events leading to independence for continence control.	Three broad themes were identified: (1) typically developing versus different: • Parents initially described child to be similar to those without disabilities, later showed that they did not see their child as a “typical” Children. • Children overwhelmingly reported that their disability inherently made them different from their peers. (2) independence, ownership and the road to continence: • Children with SB who achieved bladder continence were more independent and participated in more social functioning. • SB adversely impacted children’s social lives and led to rejection and social isolation, which in turn led to stigma and poor social functionality (3) peer relationships and acceptance: • 3 children reported many close friends and 4 reported no friends, 2 children reported being repeatedly bullied at school as a result of their disability, • Incontinence was seen as a barrier to participation and peer relationships by many parents and children.	Study type = 3 Sampling = 2 Data collection Method = 1Analysis = 3 Total score = 9
Marques et al (2015) 42 USA	Identify relationships between psychosocial variables and physical activity in children and adolescents with SB	Quantitative, cross-sectional study 31 children with SB (aged 10-17 years old)	Perception of physical activity • Questionnaire: leisure time activities (21 activities) including participation in organized sports activities • Students’ attitudes toward physical education and physical activity • Questionnaire: psychosocial variables (attitudes, sports goal orientation and perceptions	• 38.7% of the children with SB participated in both organized and non-organized physical activity. • No relationship was found between participation in physical activities and psychosocial variables. • A statistically significant positive correlation was found between perception of competence and participation in non-organized physical activity.	Study type = 3 Sampling = 2 Method = 1 Analysis = 3 Total score = 9
Holbein et al (2017) 5 USA	Examine how parents’ expectations relate to actual milestone achievement, and to compare milestone achievement in emerging adults (18-25 years) with spina bifida with that of peers with typical development.	Quantitative, experimental longitudinal study Milestone achievement in present study focuses on data collected at Time (T)2, when assessment of parental expectations began, T5 to assess changes over time, and T7 when participants were 22 to 23 years old (milestone achievement). T2: 67 families of children with SB (10-11 years old) and 66 families of children with typical development T5: 52 families (SB) (16-17 years old) and 61 (typical dev.) T7: 56 SB (22-23 years old) and 61 (typical dev.)	Parental Expectations Measure designed for this study: parent rated extent to which they believe their child capable of attaining specific adult milestones (independent living, college attendance, full-time job, marriage, and parenthood)	• Overall mean percent expectations at T2 were relatively high for each of the five milestones with the lowest expectation for likelihood of becoming parents. • Parental expectations significantly decreased from T2 to T5 for their child’s likelihood of living independently, obtaining full-time job, and getting married. • In all five adulthood milestones assessed at T7, emerging adults in the spina bifid group were significantly less likely to have achieved the milestone compared with the emerging adults with typical development.	Study type = 4 Sampling = 3 Data collection Method = 1Analysis = 3 Total score = 11
Bakaniene & Prasauskiene (2019) 26 Lithuania	Analyze patterns and predictors of social participation in Lithuanian children and adolescents with SB	Qualitative, cross- sectional study 99 parents of the children with SB (5-18 years).	Participation and Environment Measure for Children and Youth (PEM-CY) (types of activity performed in home, school, and community. Factors that influence the child’s participation such as physical layout, availability of programs, cognitive/social demands for activity)	• Children with spina bifida participated most frequently and in a higher number of activities at home (Mean participation frequency 5.75 out of 7, with 88.1% participation in all possible activities), followed by school (Mean participation frequency 3.89 out of 7, with 73.1% participation in all possible activities) and community (Mean participation frequency 2.07 out of 4.6, with 50.6% participation in all possible activities); desired changes were least reported at home (47%). • Parents wanted to see an increased number of activities at school and in the community. • Environmental factors consistently explained participation dimensions across settings, with overall environmental supports at 92.4% at home, 84.5% at school, and 75.6% in the community. • Predictors of participation in children with spina bifida included strong associations with environmental helpfulness (92.4%) and few need special educational support due their special needs (35.4%) and overall environmental support (84.5%) at school, and overall environmental support (75.6%) in the community. • Intellectual disability correlated with home-schooling as well. • Bowel incontinence and environmental barriers impacted school participation frequency; shunt revisions influenced desire for change in school activities. • Overall, environmental factors significantly contributed to explaining participation patterns across settings. • A significant barrier to community participation reported by parents was the lack of public transportation (42%)	Study type = 3 Sampling = 2 Data collection Method = 1Analysis = 3 Total score = 9
Papadakis et al (2021) 41 USA	Investigate the relationship between sociodemographic factors and various characteristics of youth with SB, including their health and their psychosocial, neuropsychological functioning	Quantitative, longitudinal study 140 youth with SB ages	Longitudinal study examining family, psychosocial, and neuropsychological functioning Condition related socioeconomic measure Health related measure Neuropsychological measure	• Youth with SB having sociodemographic risks (Ethnicity, education, health insurance, parent employment, occupation, immigrant status, language, family structure, income to need ratio) experienced greater pain but fewer urinary tract infections. • Academic performance and age difference did not affect these relationships.	Study type = 3 Sampling = 2 Method = 1 Analysis = 3 Total score = 9
Darow (2023) 34  USA	Compare longitudinal trajectories of Cognitive Disengagement Syndrome (CDS) in youth with spina bifida (SB) versus typically developing (TD) peers and link these to social skills and behavioral autonomy.	Quantitative, longitudinal cohort: 68 SB and 68 TD children (ages 8-9 at T1) assessed at five biennial time points (ages 8-17), multi-informant (mother, teacher) CBCL CDS, social skills, and decision-making questionnaires.	CDS: Three-item CBCL scale (confusion/fog, daydreams, low energy), mother & teacher report at T1–T5; Social skills: Mother, father, teacher SPPC Social Acceptance (T5); self-report SSRS (T5); Behavioral autonomy: Steinberg Decision-Making Questionnaire (T5).	• Teacher-reported CDS intercept higher in SB (1.60) vs • TD (1.02); slopes non-significant (stable trajectories). • In SB group, baseline teacher CDS predicted poorer father- (b = −0.53, P < .05) and teacher-reported social skills (b = −0.80, P < .001). • In SB group, increasing mother-reported CDS over time predicted lower mother-rated social skills (b = −0.43, P < .01) and reduced behavioral autonomy (b = −0.43, P < .01).	Study type = 4 Sampling = 2 Data collection Method = 1Analysis = 3 Total score = 10
Suhs (2025) 29 USA	Investigate how observed and self-reported dimensions of friendship relate to mental health and physical adjustment in children/adolescents with SB.	Qualitative Cross-sectional study of 140 sample with SB + their mean age 11.4 years.	Observed interaction quality: coded affect, responsiveness, engagement during a structured peer task; Friend quantity: self-report counts of friends; Self-report friendship quality: Friendship Quality Scale & emotional support items; Health outcomes: CBCL withdrawn/depressed subscale; health adjustment questionnaire.	• Higher observed interaction quality was associated with both fewer withdrawn/depressed behaviors and better physical health adjustment. • Greater number of friends correlated with lower withdrawn/depressed behaviors but did not predict physical health. • Self-reported emotional support related to better emotional quality of life only.	Study type = 4 Sampling = 2 Data collection Method = 1Analysis = 3 Total score = 10

Quality Appraisal

To establish methodological quality, each selected study was evaluated using the quality appraisal tool developed by Olsen and Baisch. ¹⁹ This tool includes 4 components: study type, sampling method, data collection method, and analysis type. The scores assigned to the study types ranged from 1 (best practice report) to 6 (quantitative experimental design). The sampling method scores were assigned as follows: 0 = not explained or NA, 1 = convenience, 2 = purposive or case-matching, and 3 = random or 100%. For the data collection method, a score of 1 or 0 was assigned depending on whether the data collection methods and tools were explained (1) or not (0). Finally, based on the highest reported level of analysis, the analysis type was scored as follows: 1 = narrative, 2 = descriptive statistics, and 3 = inferential statistics. Consequently, the total tool scores ranged from 2 to 13; the higher the total score, the better the quality of the study. ¹⁹ Quality appraisal scores are listed in Table 1.

Results

Study Sample Characteristics

Table 1 provides a detailed summary of the studies included in this review. Five qualitative and 11 quantitative studies accounted for 31% and 69% of the studies, respectively. A total of 1489 children with SB were included in this study from various different studies. Notably, it’s important to mention that some studies of children with SB also included children without SB, specifically those with SCI, and typically developing peers. A total of 510 parents participated in these studies. The sample size range of children with SB was 20 to 140, while that of the parents was 20 to 90. Child/adolescent participants ranged from 2 to 18 years of age; however, a few studies reported adult participants up to 23 years of age.

As shown in Table 1, the studies had both quantitative and qualitative research designs. Among the quantitative studies, cross-sectional, prospective, longitudinal, and experimental designs were employed. Eleven studies were quantitative in nature, while the remaining 5 were qualitative, employing phenomenological, grounded theory, descriptive, and cross-sectional designs. A wide variety of instruments have been used to measure social participation, such as the Children’s Assessment of Participation and Enjoyment (CAPE), Activities Competence Scale of the Child Behavior Checklist (CBCL), School Function Assessment (SFA), and Participation and Environment Scale for Children and Youth (PEM-CY). In addition, open-ended questions about participation, the influences of participation, and barriers to participation in social activities were used during the interviews in the qualitative studies.

Examples of instruments used to measure peer relationships include the General Friendship Interview, Didactic Friendship Interview, Friendship Quality Scale, and open-ended questions in qualitative studies about interactions with friends.

Study Quality

Study quality was assessed by calculating the appraisal scores using the Olsen and Baisch’s quality appraisal tool. ¹⁹ Each study score was calculated with 13 as the maximum score and 2 as the minimum score. The overall quality appraisal scores (Table 1) for all studies ranged from 7 to 9.

Thematic Results

Guided by models of social participation in youth with chronic health conditions by Pinquart and Teubert, ⁸ the synthesis was organized into the following domains: (1) School and Community Participation, (2) Peer Relationships and Friendships, (3) Family Interactions and Parental Role, and (4) Independence and Autonomy. This integrative review revealed several key findings: Researchers have indicated that children with SB often show limited participation in school, community, and physical activities. Overall, this review shows that children with SB face various challenges in social functioning. Parents and friends significantly influenced the lives of individuals with SB regarding the social functioning of children with SB played a pivotal role in shaping their developmental trajectory. The synthesis identified 4 domains: (1) School and Community Participation, (2) Peer Relationships and Friendships, (3) Family Interactions and Parental Role, and (4) Independence and Autonomy. The domains that emerged from the integrative synthesis are presented below:

School and Community Participation

Engaging in school, community, and physical activities can be difficult for children with spina bifida (SB). However, parents felt that participating in outdoor sports could improve their children’s social and academic abilities, as well as their general health and well-being. ²⁰

Various factors contributing to restricted social participation among children with SB were identified in this review, including mobility, incontinence, and intellectual challenges among others. Children with SB who use wheelchairs tend to have more limitations in social functioning due to difficulties with building accessibility and long-distance transportation. Fischer et al ¹² revealed that children with SB who achieved bladder continence were more independent and participated in more social functioning than children who experienced incontinence. Similarly, compared to their younger peers, older children with spina bifida (SB) engage in fewer leisure, physical, and skill-based activities. Child’s gender and level of schooling had no impact on participation. However, youth with employed caregivers participated in more social activities. Child motor level, ambulation, and bladder/bowel issues showed no significant impact on participation, except for youth without a shunt, who participated more in physically demanding and skill-based activities. The study also highlighted a link between increased pain and reduced social competence, with greater pain intensity decreasing social activity. The association between social competence and pain decreased with increasing engagement in social activities. Additionally, decreased social activity has a significant negative effect on social competence.^{12,21 -23} Another study showed that various factors affect active community engagement. Among these factors are low motivation, lack of information, time constraints, and insufficient family support. ²⁴ Similarly, Papadakis et al ⁴¹ found that in youth with SB, sociodemographic factors, including race, ethnicity, health insurance status, education level, and employment status, significantly and negatively affected academic performance and social functionality in some individuals with SB.

Family Interactions and Parental Role

Holbein et al ²⁵ reported that parents of children with Spina Bifida (SB) aged 10 to 11 held high expectations for their children to achieve 5 life milestones: independent living, college attendance, full-time employment, marriage, and parenthood. However, when their children reached ages 16 to 17, these expectations significantly decreased compared to their counterparts with typical development. Optimism, sociodemographic factors, and the child’s larger environment are some of the factors that shape the nature and impact of parental expectations. ²⁵

Researchers have found that parents are the most important source of support for children with SB to participate in social activities. In addition, parents provide a great deal of care by imparting health-related information and providing support for their children’s emotional well-being and sense of self to ensure their quality of life.^24,26

Despite parents’ strong level of support, researchers have reported that most parents believe that their children with SB differ in aspects of their social participation from children without ailments, including siblings. In one qualitative study addressing parents’ perceptions of how their children with physical disabilities were influenced by outdoor sporting activities, thematic categories emerged from interviews: achieving good health, being part of a social group, and learning a sporting activity. These categories reflect the importance parents place on sports activities for children and adolescents with physical disabilities. ²⁰ Moreover, parents lowered their expectations of their children with SB for academic and social performance over time because of their condition; however, optimistic parents showed increased expectations. ²⁰

Peer Relationships and Friendships

While this is not always the case, children and adolescents with SB are more likely to name a healthy peer as their best friend. ²⁷ Kids and teens with SB might benefit from peer support, although such assistance is often lacking. Peny-Dahlstand et al reported that although most children with SB try to explain their condition to their friends, they rarely discuss the difficulties surrounding bowel and bladder issues, which are the challenges that most interfere with their social participation. ²¹ In addition, peer acceptance of friends with SB varied considerably among studies, with some children reporting many close friends and others reporting that they had no friends.^12,27 In a study by Stiles-Shields et al, many self-reported friendship qualities did not differ markedly from peers, which mirrors Stiles-Shields et al’s longitudinal findings that friendship trajectories for many quality domains are similar for youth with SB and their chosen peers, supporting a strengths-based interpretation. ²⁸ Furthermore, Peny-Dahlstrand et al ²¹ found that 89% of individuals with SB had low levels of interest in participating in and being involved in school activities, especially structured activities in recess and playground settings. Youth with SB spend fewer days with friends and show lower levels of security, companionship, and emotional support from friends and family. ²⁷ Furthermore, Suhs et al ²⁹ examined 140 children and adolescents with spina bifida (mean age 11.4 years; 53.6% female) and their closest friends to show that observed peer-interaction quality, characterized by positive affect, responsiveness, and mutual engagement during a structured task, was significantly associated with both less withdrawn or depressed behaviors and better physical health adjustment. While self-reported emotional support was associated with enhanced emotional quality of life but not with physical health outcomes, friendship quantity, that is, the number of friends, was linked only with fewer withdrawn/depressed behaviors and did not predict physical health outcomes. ²⁹

Independence and Autonomy

In addition, for children with SB, learning and intellectual disabilities can increase their risk of not participating in social functions. ³⁰ Bakaniene and Prasauskiene ²⁶ and Bloemen et al ³¹ focused on the influence of environmental variables as they investigated the patterns and determinants of social involvement in Lithuanian children and adolescents with SB and found that social participation is mainly affected by the home and school settings where they can be encouraged to participate or negatively influenced by the contrary. Furthermore, the social participation of children with spina bifida is shaped by a confluence of elements, encompassing their physical capacities, functional capabilities, and the extent of support provided by their environment. ³⁰ These findings were further supported by Flanagan et al, who found that children with SB engaged in fewer activities than those with paraplegic SCI. ³²

Social functioning in young adults with SB can be affected by their inability to reach normal developmental milestones. Holbein et al observed that emerging adults with SB exhibited a lower likelihood of attaining milestones such as attending college, securing full-time employment, living independently, getting married, and becoming parents than individuals with typical development. ²⁵ Furthermore, even though children with SB were less likely to get a driver’s license, work in a competitive job, or hang out with friends, according to Liptak et al, 76% had competitive jobs or were enrolled in school, 30% had a driver’s license or learner’s permit, and 15% went on dates and spent time with friends. ³⁰ Murray et al ³³ caution that improving social functioning may also increase exposure to risky behaviors (eg, alcohol, sexual activity), so interventions should combine social-skill enhancement with risk-prevention education. ³³ According to the findings of Darow et al, ³⁴ the levels of Cognitive Disengagement Syndrome (CDS) that were judged by teachers at the beginning of the study were substantially higher in pupils with SB compared to their classmates with Typically developing (TD) (intercepts: SB = 1.60 vs TD = 1.02). However, the intercepts rated by mothers did not vary significantly (SB = 1.03; TD = 1.05). According to growth curve analysis, the CDS was constant throughout the 5 evaluations. More importantly, in the SB group, higher baseline teacher-reported CDS predicted lower father-rated (b = −0.53) and teacher-rated social skills (b = −0.80), and larger increases in teacher-reported CDS over time further predicted eventual social skill deficits (b = −0.54). Moreover, among SB kids, increases in mother-reported CDS specifically predicted worse behavioral autonomy (b = −0.43). These longitudinal correlations highlight how much social functioning and decision-making autonomy in children with spina bifida are undermined by both the degree and trajectory of attentional disengagement. ³⁴

Children with Spina Bifida exhibit incredible resilience and a tremendous desire to participate in their communities despite the challenges they encounter. ³⁰

Discussion

Our research provides important new information on the social functioning of children with SB. First, when comparing children and adolescents with SB to those with other chronic diseases, researchers have consistently shown poorer social functioning in patients with SB. However, the degree of impairment varied across the reviewed studies, underscoring important similarities and differences. Similarities across studies included the repeated identification of incontinence, hydrocephalus, mobility challenges, and cognitive delays as primary barriers to social participation. Regardless of geographic or cultural context, continence problems were strongly associated with isolation, reduced self-esteem, and withdrawal from peer activities. Similarly, motor impairments and executive dysfunctions were recurrently linked to limited participation in physical and school activities. Differences among studies largely reflected contextual and methodological variations. For instance, studies from high-income countries frequently emphasized environmental modifications, inclusive education, and structured rehabilitation programs as protective factors, while research from low- and middle-income settings highlighted the amplifying effects of poverty, stigma, and inadequate healthcare services. Qualitative studies, especially those incorporating children’s voices, described nuanced experiences of shame, resilience, and identity formation that were less visible in quantitative surveys. By contrast, quantitative studies often relied on standardized instruments (eg, PedsQL, SOC scales), which facilitated cross-country comparisons but may not fully capture cultural or contextual differences in lived experiences. Another divergence lay in the treatment of parental expectations. Some studies reported that parents of children with SB lowered milestone expectations over time, whereas others highlighted the risks of overprotective attitudes that restricted independence.

This highlights the unique social issues encountered by people with SB.

We investigated how parental expectations affect the social engagement of children with SB. It is important to recognize the complex interactions between parental expectations and the social functioning of children with SB, ³⁵ even though the specific findings pertaining to these expectations were not clear in the summaries provided. A more thorough analysis of how parental expectations either exacerbate or lessen barriers to social participation for children with SB may be beneficial for future studies and interventions. ³⁶

The identified lower social functionality in children and adolescents with SB compared to those with other chronic conditions underscores a significant challenge in their interactions with friends and family members. ³⁰ This limitation appears to be mainly related to the lack of facilities that meet the requirements of children with chronic illnesses, such as SB, and parents’ ignorance of the existence of these facilities. ³⁷ This highlights a crucial gap in the systems of support for children with SB, which impedes their ability to interact socially and might even make them feel alone. To address this complex issue, comprehensive initiatives are required to improve infrastructure and public knowledge of the resources available for children with SB and their families.

This review demonstrated that several factors significantly limit the social participation of children with SB. Incontinence issues lead to isolation, and motor level restrictions hinder engagement in activities such as running or jumping.^5,12 Cognitive delays associated with hydrocephalus pose additional challenges. ⁹ Parental employment and income levels can affect access to social opportunities. Despite mothers being the primary source of support, there has been limited exploration of how parental expectations influence the social functioning of children with SB. The impact of incontinence on children’s social participation was evident, leading to isolation and restriction of engagement with others. Incontinence is considered a major health issue for children, as some tend to hide it from their families and friends. It is crucial to understand the factors that affect children with SB to participate in social development, which could help children who need intervention. Motor level restricts children’s ability to engage in activities, specifically those that require running or jumping skills. Children with hydrocephalus tend to experience cognitive delays that affect their participation in sports. Parents’ employment was related to social participation because income may provide more opportunities for children to engage in private centers or extended networks. Furthermore, although some investigators reported that mothers served as the primary source of support for children with SB, a notable gap identified in this review is the absence of studies explicitly connecting parental expectations to social functioning in SB, despite robust evidence for such links in academic and developmental contexts. This absence is itself important: it highlights a critical area for future inquiry and suggests that parental expectations, while well-established as predictors in other pediatric domains, remain underexplored in SB. Based on the literature, children born with disabilities are viewed as vulnerable by their parents, especially when compared to their healthy peers or siblings. This viewpoint may lead to overprotective behavior on the part of parents, which can, in turn, result in less functioning in a child. According to Holmbeck et al, ³⁸ parents of children with SB showed greater overprotective behavior than parents of children without the condition, and children of overprotective parents may have less autonomy, less social maturity, and a limited ability to interact with other children. ³⁸ It is often not emphasized or researched in detail which parent views the child with SB more vulnerable that is, father or mother. Kritikos et al examined informant discrepancies between mother and father reports of child vulnerability in 92 families of youth with spina bifida. They found that fathers were more likely than mothers to rate their child as highly vulnerable, and that these “father-high/mother-low” discrepancies were significantly associated with greater paternal mental-health symptoms, higher parenting stress, poorer parenting alliance, and more child behavior problems. The authors conclude that such father–mother perceptual gaps may serve as a “red flag” for clinicians, signaling the need to assess parental adjustment and family functioning. ³⁹ According to Holbein et al, parents of children with spina bifida decreased their expectations for the majority of milestones with time; these expectations were positive and had nothing to do with the actual attainment of milestones. Compared to those with regular development, emerging adults with spina bifida had a lower chance of reaching every milestone. While it’s crucial for families to establish reasonable objectives, children with spina bifida and their families may benefit from optimistic parental expectations. ²⁵

The effects of spina bifida on several bodily systems, such as the neurological, musculoskeletal, genitourinary, and gastrointestinal systems, as well as the influence on a child’s psychosocial development, need multidisciplinary treatment. Therefore, the findings of this review underscore the importance of interdisciplinary care in developing interventions to maximize the social functioning of adolescents with SB, regardless of their physical limitations. Future intervention research of interdisciplinary teams that assist parents with their ability to help their children with spina bifida engage in social functioning and become more socially involved with family and friends is needed. Furthermore, social activities such as sleepovers, camp overnights, dating, and social and recreational activities outside the home should be encouraged. This could help in development and maintenance of friendships. ⁴⁰ Such research could reveal avenues for parents and other caregivers to empower children and adolescents with SB, helping them better understand and accept their bodies and take pride in their identity, rather than viewing themselves as broken or shameful. ¹² In addition, the limited body of research on the association between incontinence and social participation should be expanded, and nurses should familiarize themselves with such research so that they can help children with SB to avoid social isolation. Healthcare providers, including nurses, can assist families in choosing to learn clean intermittent catheterization, which can reduce incontinence and influence social participation.

Future Implications

This review highlights the need for multidisciplinary interventions that go beyond physical rehabilitation to include cognitive, psychosocial, and educational supports for children with spina bifida. A key gap is the limited evidence on parental expectations, which future studies should explore through longitudinal and intervention-based designs to determine how realistic yet optimistic outlooks influence social outcomes. Rigorous, prospective SB studies using multi-informant measures and standardized social outcomes are needed to clarify whether and how expectations shape social functioning. Greater attention to environmental supports such as accessible infrastructure, inclusive school policies, and stigma reduction is also critical. Finally, programs that prepare families and youth for the transition to adolescence and adulthood, combining social-skill building with risk-prevention strategies, may help maximize participation and independence.

Limitations

Despite the multiple insights reported, this review has limitations. First, the review focused only on studies published in English; therefore, relevant studies published in other languages may have been omitted. Second, the quality appraisal performed for this review found that the quality scores were lower for 9 studies because they did not fully address their sampling and data collection methods; this limitation reduced the significance of their findings and conclusions. In contrast, the 7 studies that detailed their research methods received overall quality appraisal scores of 8 and 9, suggesting that their findings can be considered more reliable and meaningful.

Conclusion

This review identified 4 domains which are engagement in social activities, parents’ expectations or roles, and peer relationships, and found consistent participation limitations among children with spina bifida. Major drivers were continence and mobility problems, hydrocephalus and neuropsychological deficits (eg, attention/executive dysfunction), and contextual barriers (stigma, limited school/community supports). We recommend interdisciplinary care that pairs medical/rehabilitation management with routine cognitive screening, psychosocial support, and school accommodations, and that includes risk-prevention education. While this review confirms consistent limitations in social functioning among children with SB, it also reveals a striking gap: the lack of studies examining the influence of parental expectations on social outcomes. Given the established role of expectations in shaping academic achievement and self-management in chronic conditions, this omission should be a priority for future research. Recognizing and addressing this gap can help families and healthcare providers develop interventions that better support children’s social integration. Thus, future nursing research should focus on addressing this gap, with the goal of maximizing the social functioning of children and adolescents with SB.