Barriers to healthcare transition for spina bifida patients: a systematic review.

OBJECTIVE: Patients with spina bifida (SB) were historically followed by pediatric providers throughout their entire lives. Through medical and surgical advancements, now more pediatric SB patients are living well into adulthood. Nonetheless, many patients fail to successfully transition to appropriate adult healthcare providers. The goal of this study was to identify factors that helped facilitate or hinder the successful transition of adolescent and young adult (AYA) SB patients to adult providers. METHODS: A systematic review was conducted exploring the transition care of SB patients using the PubMed, Embase, and Scopus databases. Titles and abstracts from articles identified were read and selected for full-text review. Studies meeting the inclusion criteria were reviewed in full and analyzed for study design, populations, interventions, and factors influencing transition. RESULTS: The primary search identified 2050 articles, of which 20 were included in the final review. Thirteen studies discussed factors relating to neurosurgical care, 8 referenced gastrointestinal and genitourinary considerations, 11 examined cognitive and psychosocial factors, and 17 explored healthcare system factors. Several barriers were consistently reported regarding communication, patient and parental attitudes and perceptions, and failure to embrace formalized and transparent protocols. Conflicting results were reported regarding the influence medical comorbidities had on a patient's ability to transition. CONCLUSIONS: The process of transitioning AYA SB patients to adult care is complex, involving an interplay of structural and psychosocial factors. The findings in this review suggest that some barriers can be alleviated with improved education, planning, and awareness of factors that influence transition care.

Commentary- Bridging the Gap: The Impact of Bowel Management Programs on Daily Life in Pediatric Colorectal Disease.

BACKGROUND: This commentary discusses the social impact of bowel management programs (BMPs) on children with colorectal diseases, including anorectal malformations (ARM), Hirschsprung disease (HD), functional constipation (FC), and spina bifida. Previous studies focused on functional outcomes, but this study bridges the gap to daily life experiences. METHODS: The study examined children's experiences in BMPs, focusing on school participation, vacation ability, and overall patient experience. Cleanliness, defined as fewer than one stool soiling episode per week, was achieved by 70% of participants. RESULTS: Positive patient experiences were linked to achieving stool cleanliness, regardless of the management method. Invasive methods like enemas did not negatively affect experiences if cleanliness was maintained. Validated patient-reported experience measures (PREMs) and patient-reported outcomes measures (PROMs) were used, though the median age of 8.9 years posed limitations. CONCLUSION: The commentary highlights the significance of stool cleanliness in improving patient experiences and supports the effectiveness of various BMP methods. Future research should include longitudinal follow-ups to assess BMP durability and gather data from older children.

Quality of Life and Its Associated Factors Among Children with Spina Bifida in Ethiopia: A Cross-Sectional Study to Inform Policy and Practice.

BACKGROUND: Congenital myelomeningocele, or spina bifida (SB), is the predominant congenital anomaly of the central nervous system. Beyond its implications on neonatal mortality, SB impacts the long-term quality of life in affected children. This study sought to investigate the health-related quality of life (HRQoL) among children with SB treated at Ethiopia's leading pediatric neurosurgical facility. METHODS: Set at Zewditu Memorial Hospital in Addis Ababa, Ethiopia, this hospital-based cross-sectional study spanned from June 30 to September 30, 2022. It incorporated 232 children, using data gathered through interviewer-led questionnaires. The HRQoL was measured using the PedsQL 4.0, a 23-item generic scale. RESULTS: The study's participants had a median age of 5 years (interquartile range = 3 to 6 years). The aggregate mean scores on the PedsQL 4.0 tallied at 68.59 ± 18.01. The lowest scores emerged from queries on school participation, whereas physical and emotional functioning registered the highest scores. Through multiple regression analyses, variables such as family income, monthly household income, number of children, and the presence of a neurogenic bladder showed strong association with HRQoL. CONCLUSIONS: This study fills a gap in the literature providing information on the HRQoL and its associated factors for children with SB in low-resourced settings. We champion the proactive integration of quality-of-life metrics into neurosurgical care policy and practice. Given the enduring consequences of SB, interventions honing the HRQoL can steer children toward realizing their intrinsic and enhance societal participation and contribution.

"Crafting a 'TransitionOmeter': A Proposed Framework for Developing and Honing Capabilities of Young People Transitioning to Adult Healthcare Services."

This paper focuses on an innovative approach to preparing children and young people, with Spina Bifida, to move from child-centered to adult-oriented healthcare systems. Reflecting on our role in delivering a national nurse led service, we set to identify and critique international transition tools in use for this population. Specifically, we aimed to identify the core capabilities and indicators of progression to successful transition, so that holistic interventions could be planned to match the needs of individuals and their carers. There were two phases to the study, initially focusing on a systematic literature review on transition tools and the specific items that these tools captured, including skills, abilities and behaviors. Phase two culminated in the articulation of a segmented and incremental "road map", aligned with facets deemed essential for a successful healthcare transition. The reporting of the literature review (phase one) followed the PRISMA guidelines and shaped the qualitative element of the study (phase two) through the use of semi-structured interviews and thematic analysis. The search strategy yielded 11 studies, which were then manually searched for other relevant literature, adding a further 14 articles. The review analyzed 7 specific tools for spina bifida and 8 generic tools, which were deemed appropriate for this group of patients. A comprehensive list of core capabilities was then articulated and framed to fit a progression timeline. Specific interventions were formulated to explore ways to co-produce resources that could enhance and support a planned transition to adult-focused services. Our proposed mapping of capabilities and progression could shape other transition programs, where nurses work collaboratively with young people, carers and other members of a team. More work is needed to further explore and embed the framework that, as we did, could be digitized and shared with all stakeholders involved in the transition process.

Linguistic, Content and Face Validity of the Swedish Version of a Quality-of-Life Assessment for Children, Teenagers and Adults with Spina Bifida.

Spina bifida includes a spectrum of different neural tube defects. Myelomeningocele is the most serious type and is associated with a risk of paralysis and sensory dysfunction below the affected level, bladder/bowel dysfunction, brain dysmorphology, and impaired health-related quality of life (HRQoL). The aim of this study was to describe the establishment of linguistic, content and face validity of the Swedish version of a Quality-of-Life Assessment for children (QUALAS-C, n = 10 items), teenagers (QUALAS-T, n = 10 items) and adults with spina bifida (QUALAS-A, n = 15 items) based on the original US English versions. The process included close collaboration with the original instrument developer and complied with international standards on patient-reported outcome measurements. The procedure includes forward translation, expert and patient/parent review and reconciliation, back translation, back translation review and cognitive debriefing interviews with 16 people with spina bifida aged 8 to 33, providing them with the possibility of evaluating the clarity, adequacy, and comprehensiveness of QUALAS-C, QUALAS-T and QUALAS-A, respectively. The interviews lasted a median of 15 min (range 8-16) for QUALAS-C, 10 min (range 9-15) for QUALAS-T and 24 min (range 9-38) for QUALAS-A. Four main issues/topics needed attention and discussion after both the forward and back translation. Following the back translation review, all issues were resolved. The patient feedback revealed recognition of the HRQoL issues included in QUALAS, and also difficulties in understanding some questions. After the patients' evaluation, four items were reworded for clarity. No study participant reported a wish to add to or remove questions from QUALAS. Hence, the Swedish versions of QUALAS became conceptually equivalent to the original US English versions and achieved linguistic, content and face validity. While empowering the voices of people with spina bifida, these results also enable their HRQoL to be properly assessed in research and clinical care in Sweden and in international studies.

Trajectories of self-management and independence in youth with spina bifida: Family-related predictors of growth.

The purpose of this study was to assess family-related predictors of self-management trajectories in youth with spina bifida (SB). Participants with SB completed the Adolescent/Young Adult Self-Management and Independence Scale (AMIS II) interview across four time points. Family functioning, family-related stress, and perceived family support were assessed by multiple reporters and multiple methods. Growth in AMIS II total self-management and the AMIS II subscales (Condition and Independent Living) were estimated using linear mixed effect models as a function of family factors, after controlling for socio-demographic, condition-related, and neuropsychological variables that had been found to be significant predictors of self-management in prior studies. Model fit and parsimony were assessed using Akaike's information criterion (AIC). This diverse community sample included 99 respondents aged 18-27 years old. About half were female (52.5%) and White (52.5%); 15.2% were Black, and 32.3% were Hispanic/Latino. Observed family cohesion at baseline was associated with all self-management scales at age 18 (all p < 0.05). Growth in self-management was associated with parent-reported number of family stress events. For growth in total self-management, the best model included age, race/ethnicity, family income, shunt status, lesion level, neuropsychological function, observed family cohesion, and an age-by-number of family stress events interaction effect. The study findings suggested that family factors were important predictors of self-management trajectories, even after controlling for socio-demographic, condition-related, and neuropsychological covariates. Risk and protective factors identified in families of youth with SB can inform family-focused interventions for self-management.

Pain Scores after Open Fetal Spina Bifida Repair and Caesarean Section: A Longitudinal Cohort Study.

INTRODUCTION: In fetal surgery, successful pain management is crucial for postoperative mobilization, prophylaxis of contractions, and fast recovery. This study analyzed patient's pain experience after open fetal spina bifida (fSB) repair in comparison to pain scores after the subsequent Caesarean section (C-section). MATERIALS AND METHODS: Data were collected with a questionnaire given to 91 women, who had fSB repair and then C-section at our center between 2019 and 2022. It comprised 12 questions covering different aspects of pain experience and satisfaction with pain therapy and was answered by 67 women after fSB repair and 53 after C-section. Postoperative pain was rated on a Likert scale from 0 (slight/rarely) to 100 (strongest/always). Outcomes after fSB repair were compared to those after C-section. Additionally, subgroup analysis compared outcomes of women with different pain levels (group 1-5) after fSB repair. RESULTS: Compared to women after C-section women after fSB repair reported significantly higher maximum pain scores (MPS) (p = 0.03), higher sleep disturbance due to pain (p = 0.03), and sedation rates (p = 0.001) as side effect from pain therapy. No differences were found regarding feelings of insecurity (p = 0.20) or helplessness (p = 0.40), as well as involvement in (p = 0.3) and satisfaction with pain therapy (p = 0.5). Subgroup analysis revealed that women with higher MPS after fSB repair were significantly more often non-Caucasians (p = 0.003) and more often affected by pain while lying in bed (p = 0.007) and during mobilization (p = 0.005). Additionally, they reported higher rates of dizziness (p = 0.02) and lower satisfaction rates with pain therapy (p = 0.03). Postoperative complication rate did not differ among groups. CONCLUSION: Although women after fSB repair reported higher MPS compared to after C-section, the current pain management was generally perceived as satisfactory.

Growth of condition-related knowledge among youth with spina bifida: associations with neurocognitive functioning and self-management skills.

OBJECTIVE: This study aims to characterize the growth in condition-related knowledge in youth with spina bifida (SB), identify neurocognitive predictors of growth, and examine associations between growth in knowledge and subsequent levels of medical self-management skills. METHODS: Participants were recruited from a larger longitudinal study involving 140 youth with SB and caregivers, who completed questionnaires and interviews every 2 years over 8 years. The current study included the youth report of condition-related knowledge and medical self-management skills. Youth attention and executive functioning were assessed via parent and teacher reports and performance-based assessment. Latent growth curves were conducted in Mplus Version 8 (Múthen, L. K., & Múthen, B. O. [1998]. Mplus User's Guide. [Eighth]. Muthén & Muthén) to examine change over time in youth-reported condition-related knowledge. Neurocognitive variables were included as predictors of growth in knowledge and regression analyses were used to predict medical self-management skills from growth in condition-related knowledge. RESULTS: Youth condition-related knowledge increased linearly. Better youth performance on working memory and attention performance-based tasks predicted a higher intercept for condition-related knowledge at T1, but not slope. Teacher and parent reports of inattention and executive dysfunction were not consistent predictors of intercept and growth. Slope of condition-related knowledge was not predictive of subsequent youth self-management skills. CONCLUSIONS: Youth with SB gain condition-related knowledge over time. However, executive dysfunction and inattention may impede gains in condition-related knowledge. Thus, executive functioning supports, attention-related interventions, and psychoeducation may support condition-related knowledge gains and later medical self-management skills, but further research assessing family and cultural factors is needed.

Thalamic connectivity topography in newborns with spina bifida: association with neurological functional level but not developmental outcome at 2 years.

Spina bifida affects spinal cord and cerebral development, leading to motor and cognitive delay. We investigated whether there are associations between thalamocortical connectivity topography, neurological function, and developmental outcomes in open spina bifida. Diffusion tensor MRI was used to assess thalamocortical connectivity in 44 newborns with open spina bifida who underwent prenatal surgical repair. We quantified the volume of clusters formed based on the strongest probabilistic connectivity to the frontal, parietal, and temporal cortex. Developmental outcomes were assessed using the Bayley III Scales, while the functional level of the lesion was assessed by neurological examination at 2 years of age. Higher functional level was associated with smaller thalamo-parietal, while lower functional level was associated with smaller thalamo-temporal connectivity clusters (Bonferroni-corrected P < 0.05). Lower functional levels were associated with weaker thalamic temporal connectivity, particularly in the ventrolateral and ventral anterior nuclei. No associations were found between thalamocortical connectivity and developmental outcomes. Our findings suggest that altered thalamocortical circuitry development in open spina bifida may contribute to impaired lower extremity function, impacting motor function and independent ambulation. We hypothesize that the neurologic function might not merely be caused by the spinal cord lesion, but further impacted by the disruption of cerebral neuronal circuitry.

Cognitive disengagement syndrome in pediatric spina bifida.

The primary aim of this study was to characterize Cognitive Disengagement Syndrome (CDS) symptomatology in youth with spina bifida (SB). One hundred and sixty-nine patients aged 5-19 years old were drawn from clinical cases seen through a multidisciplinary outpatient SB clinic at a children's hospital between 2017 and 2019. Parent-reported CDS and inattention were measured using Penny's Sluggish Cognitive Tempo Scale and the Vanderbilt ADHD Rating Scale. Self-reported internalizing symptoms were measured with the 25-item Revised Children's Anxiety and Depression Scale (RCADS-25). We replicated Penny's proposed 3-factor structure of CDS with slow, sleepy, and daydreamer components. The slow component of CDS overlapped heavily with inattention, while the sleepy and daydreamer components were distinct from inattention and internalizing symptoms. Eighteen percent (22 of 122) of the full sample met criteria for elevated CDS, and 39% (9 of 22) of those patients did not meet criteria for elevated inattention. Diagnosis of myelomeningocele and presence of a shunt were associated with greater CDS symptoms. CDS can be measured reliably in youth with SB and can be discriminated from inattention and internalizing symptoms in this population. ADHD rating scale measures fail to identify a substantial portion of the SB population with attention-related challenges. Standard screening for CDS symptoms in SB clinics may be important to help identify clinically impairing symptoms and design targeted treatment plans.

Comparing Cognitive Disengagement Syndrome Growth in Youth With and Without Spina Bifida.

OBJECTIVE: Cognitive disengagement syndrome (CDS; formally known as sluggish cognitive tempo), difficulties with social engagement, and lower levels of autonomy have been identified as maladaptive comorbidities in youth with spina bifida (SB). This study compared growth curves of CDS for youth with and without SB and examined whether these trajectories were associated with later functioning. METHODS: Longitudinal data spanning 8 years included youth with SB (n = 68, Mage = 8.34) and a demographically matched sample of typically developing (TD) peers (n = 68, Mage = 8.49). Adolescents, along with their caregivers and teachers, reported on youth social skills, behavioral functioning, and CDS. Growth curve models were examined by comparing CDS trajectories by SB status. RESULTS: Growth curves indicated that youth with SB had higher levels of teacher-reported CDS at ages 8 and 9, but growth curves were relatively stable for both groups. When predicting social skills, higher levels of teacher-reported (but not mother-reported) CDS at baseline predicted worse social functioning for both youth with and without SB in adolescence. For the slope findings, higher rates of mother-reported CDS over time predicted worse social skills (ß = -0.43) and lower levels of youth decision-making (ß = -0.43) for the SB group, while higher rates of teacher-reported CDS predicted worse social skills for the TD group. CONCLUSION: Next steps include understanding the impact that impaired social functioning and restricted autonomy have on youth with and without SB due to CDS to inform interventions. Additionally, advocacy for increased awareness of CDS-related impairment is needed, particularly for youth with chronic health conditions.

Foot deformity and quality of life among independently ambulating children with spina bifida in South Korea.

BACKGROUND: Children with spina bifida (SB) may have congenital or acquired foot deformities due to neurological defects in the spinal cord. As the musculoskeletal system keeps growing, foot deformities can develop or become aggravated. Thus, healthcare providers should provide constant monitoring and proper orthopedic management. Since foot deformities can affect not only the gait but also the daily life of children with SB, it is necessary to investigate the impact of foot deformities on everyday life. The purpose of this study was to examine the relationship between foot deformity and health-related quality of life (HRQoL) among independently ambulating children with SB. METHODS: This cross-sectional study examined the associations between foot deformity and HRQoL using two patient-reported outcome measures (Oxford Ankle Foot Questionnaire, Pediatric Outcomes Data Collection Instrument) in 93 children with SB aged 7-18 years between January 2020 and July 2021. RESULTS: Children with foot deformity (n = 54) reported lower scores in all subscales (physical, school and play, emotional, and footwear) of the Oxford Ankle Foot Questionnaire for children than those without foot deformity (n = 39; p < 0.001). Additionally, in terms of the Pediatric Outcomes Data Collection Instrument, children with foot deformity also reported poorer scores in four subscales (transfer and basic mobility, sports and physical functioning, comfort and pain, happiness with physical functioning; p < 0.001) than those without foot deformity, whereas upper extremity functioning was not significantly affected. Children with foot deformities, particularly those with bilateral foot deformities, equinus deformities, or mixed deformities, which are different types of right and left foot deformities, have a lower perceived HRQoL (p < 0.05). CONCLUSIONS: Among independently ambulating children with SB, those with foot deformities showed lower HRQoL. Moreover, children with foot deformities tend to have other clinical problems, including bladder and bowel dysfunction. Therefore, orthopedic management should consider the multifaceted factors that affect children's daily life and HRQoL.

Codevelopment of an illustration representative of people living with spina bifida for health educational materials.

BACKGROUND: The lack of health education resources specific to people with disabilities contributes to disparities in outcomes. Developing user-centered materials with representative images tailored to people with disabilities could help improve knowledge and outcomes. OBJECTIVE: As a first step in developing an online sexual health resource for adolescents with physical disabilities, we sought end-user feedback to create illustrated characters for use in educational materials. METHODS: Two styles of characters were developed by the research team, which included a professional disability artist. Verbal and online survey feedback was obtained at the Spina Bifida Association's Clinical Care Conference. A new image was created incorporating initial feedback. The new image and favored image from the first round were then tested through an online survey advertised on the Spina Bifida Association's Instagram story feed. Open-ended comments were organized by categories and overlapping themes. RESULTS: Feedback was obtained from 139 audience members and 25 survey respondents from the conference and 156 Instagram survey respondents. Themes included depiction of disability, nondisability diversity, other physical appearance, emotional response, and design style. Most frequently, participants suggested the inclusion of characters with a range of accurately depicted mobility aids and of characters without mobility aids. Participants also wanted a larger, more diverse group of happy, strong people of all ages. CONCLUSIONS: This work culminated in the codevelopment of an illustration that represents how people impacted by spina bifida view themselves and their community. We anticipate that using these images in educational materials will improve their acceptance and effectiveness.

A patient- and parent-centered approach to urinary and fecal incontinence in children and adolescents with spina bifida: understanding experiences in the context of other competing care issues.

INTRODUCTION: Despite significant and known challenges to urinary and fecal incontinence (UI and FI, respectively) among children with spina bifida (SB) and their families, few studies have identified patient-centered measures and approaches to assessing them. This study represents the first stage of a larger study to develop a patient-centered goal-setting tool to guide incontinence management (Figure). Our aim was to understand patient experiences of UI and FI, and what goals should be included in the tool. METHODS: We used a qualitative research approach integrated with human-centered design methods. We recruited, in clinic and online, children with SB (8-17 years old) and parents (>=18 years old) of children with SB (8-17 years old). Online activities were analyzed by four experienced design researchers using affinity diagramming, group analysis and modeling activities (mind maps, challenge maps, experience maps). Recruitment and thematic qualitative analysis continued until saturation was reached. RESULTS: Seventeen children with SB participated (9 female, 12 shunted, 13 using bladder catheterizations, 6 using Malone antegrade continence enema, median age: 15 years old). Fifteen parents participated (13 mothers/2 fathers, median age: 42 years old), including six mother-child dyads. Five major themes each were identified for UI and FI experiences: (1) negative emotional impact of incontinence, (2) unpredictability of incontinence, (3) challenging/unpleasant incontinence management, (4) inconvenient/unreliable incontinence management, (5) UI management having unpleasant complications and FI putting much responsibility on parents. We identified six UI goal domains and five FI goal domains. Four overlapping domains included: accidents, independence, interruptions at school and social/friends. Unique domains were: urinary tract infections (UI), catheterizations (UI) and enemas (FI). COMMENT: Findings of our study improve our understanding of children's and parents' experiences associated with incontinence in SB and potential continence goals. CONCLUSIONS: Children with SB experience incontinence as distressing and unpredictable. Incontinence management can be challenging and unreliable. Patient-centered continence goals cluster in domains, indicating how incontinence intermeshes with other life areas and offering potential approaches to structure continence goals in a goals-selection tool.

Caregiver Expressed Emotion in Families of Youth With Spina Bifida: Demographic, Medical, and Family Correlates.

OBJECTIVE: Caregiver expressed emotion (EE), an interview-based measure of emotional valence within an interpersonal relationship, is associated with psychosocial outcomes across multiple conditions. Guided by a model implicating a bidirectional role of "Chronic Family Stress" in the unfolding of EE in family environments, the current study examined demographic, medical, and family-level variables in association with EE in caregivers of children with spina bifida (SB). METHODS: Data were combined from 2 distinct studies of families with a child with SB, resulting in a sample of 174 (ages 8-17). Linear regressions examined the family stressors and child variables in association with maternal and paternal warmth and criticism, as coded from EE interviews. RESULTS: Higher levels of family stress were associated with paternal criticism (p = .03), while having non-Hispanic White children was associated with both maternal and paternal criticism (ps < .005). Having children younger in age (ps < .01) and without a shunt (ps < .01) was associated with higher warmth. CONCLUSIONS: Family stressors, absence of the negative impacts of systemic racism, shunt status, and age appear to be associated with the expression of EE in caregivers of a child with SB. Findings highlight multiple assessment considerations, including assessing EE when children are younger to engage caregivers with children with SB when they are more likely to be expressing more warmth. Pinpointing factors associated with caregiver EE in SB will help to better identify families at risk for high levels of criticism and also aid in the development of targeted prevention and intervention programs.

Education and employment as young adults living with spina bifida transition to adulthood in the USA: A study of the National Spina Bifida Patient Registry.

AIM: To describe the education and employment transition experience of young adults with spina bifida (YASB) and investigate factors associated with employment. METHOD: We queried education and employment data from the US National Spina Bifida Patient Registry from 2009 to 2019. We applied generalized estimating equations models to analyze sociodemographic and disease-related factors associated with employment. RESULTS: A total of 1909 participants (850 males, 1059 females) aged 18 to 26 years contributed 4379 annual visits. Nearly 84% had myelomeningocele and, at last visit, the median age was 21 years (mean 21 years 5 months, SD 2 years 10 months). A total of 41.8% had at least some post-high school education, and 23.9% were employed. In a multivariable regression model, employment was significantly associated with education level, lower extremity functional level, bowel continence, insurance, and history of non-shunt surgery. This large, national sample of YASB demonstrated low rates of post-secondary education attainment and employment and several potentially modifiable factors associated with employment. INTERPRETATION: Specific sociodemographic, medical, and functional factors associated with employment are important for clinicians to consider when facilitating transition for YASB into adulthood. Additional research is needed to understand the impact of cognitive functioning and social determinants of health on transition success in YASB. WHAT THIS PAPER ADDS: There were low education attainment and employment rates in a large sample of young adults with spina bifida. Specific sociodemographic, medical, and functional factors are associated with employment. Some employment-associated factors, such as continence and self-management skills, are modifiable.

Solo and Partnered Sexual Behavior Among an International Sample of Adults With Spina Bifida.

BACKGROUND: Spina bifida (SB) may differentially impact adults' participation in solo and partnered sexual behaviors, but little research investigates this topic. AIM: Describe solo and partnered sexual behaviors among an international sample of adult men and women with SB. MAIN OUTCOME MEASURES: Ever participated (no/yes) and recent participation (>1 year ago/within last year) in solo masturbation, cuddled with a partner, held hands with a partner, kissed a partner, touched a partner's genital, had genitals touched by a partner, gave a partner oral sex, received oral sex from a partner, vaginal sex, anal sex, and sex toy use. METHODS: Data were drawn from a larger cross-sectional, internet-based survey assessing the sexual behaviors of an international sample of men and women with SB. We used logistic regression to examine the impact of background (gender, age, independent living, and relationship status) and health (shunt status, ambulation, and genital sensation) factors on each outcome. RESULTS: The sample consisted of 345 respondents aged 18-73 years from 26 nations. Very few (<3%) had no lifetime experience with any solo or partnered behaviors; 25.0% reported participating in all behaviors at some point in their lives. The median number of past year sexual behaviors (of 16 total) was 7. Lifetime and recent participation were associated with demographic and health factors. CLINICAL IMPLICATIONS: Despite impairment, adults with spina bifida do participate in solo and partnered sexual behaviors. Medical personnel who work with this population should include discussions about sexuality as part of routine care. STRENGTHS & LIMITATIONS: Although this research measured solo and partnered sexual behavior in large international sample of adults with spina bifida, it is limited by its cross-sectional retrospective design and non-clinical convenience sample. CONCLUSION: Despite disability, many adults with SB participate in solo and partnered sexual behavior. Medical and psychosocial supports are needed to help adults in this population enjoy sexuality in a healthy and safe manner. Hensel DJ, Misseri R, Wiener JS, et al. Solo and Partnered Sexual Behavior Among an International Sample of Adults With Spina Bifida. J Sex Med 2022;19:1766-1777.

Nurse-led eHealth transition care program for adolescents with spina bifida: A feasibility and acceptability study.

PURPOSE: This study aimed to evaluate the feasibility and acceptability of a nurse-led eHealth transition care program for adolescents with spina bifida. DESIGN AND METHODS: This study used a single-arm, pretest-posttest intervention study. Adolescents with spina bifida, aged 12-15 years, and their parents participated in the program. A 6-week program was delivered through an online platform in real-time by nurses. We evaluated feasibility and acceptability using criteria such as the completion rate, program satisfaction, changes in transition readiness, social support, career preparation behavior, sexual knowledge, and sexual worries at three time points from July to September 2021. RESULTS: Thirteen adolescents completed all sessions and surveys (13/14, 92.9%). All adolescents expressed high satisfaction with both the content and delivery methods of the program. Significant benefits in transition readiness, career preparation behavior, and sexual knowledge were identified over the study period. However, the evaluation of social support and sexual worries did not demonstrate any significant improvements. Additionally, through family counseling, adolescents benefited from experiences such as reflecting on their current transition readiness, setting and achieving individualized goals and plans using a self-checklist with their parents and nursing professionals. CONCLUSION: This nurse-led eHealth intervention was feasible and acceptable for adolescents with spina bifida. Furthermore, our results highlight the practicability and the potential for strategic dissemination of using this eHealth program in transitional care during the COVID-19 pandemic. PRACTICE IMPLICATIONS: The eHealth transition care program contributes to broadening existing nursing interventions not only in medical areas but also in daily life areas.

Trajectories of Marital Satisfaction among Parents of Youth with Spina Bifida.

OBJECTIVE: This study examined marital satisfaction among parents of youth with spina bifida (SB) over an 8-year period and investigated the usefulness of the double ABCX model for understanding factors that predict change in marital satisfaction across child age. METHODS: Data from five time points of a longitudinal investigation of psychosocial outcomes in youth with SB were included. Mothers and fathers of children with SB (aged 8-17) reported on marital satisfaction and components of the double ABCX model. Change in marital satisfaction was examined across child age with components of the double ABCX model, as well as interactions between components, as predictors. RESULTS: Marital satisfaction was significantly higher in the present sample than in a normative sample of married couples. Although there were no significant changes in marital satisfaction for either parent as a function of child age, there was significant variability for the intercept and slope of maternal and paternal marital satisfaction within the sample. Family support predicted a higher intercept, and mental health symptoms predicted a lower intercept, for maternal and paternal marital satisfaction. More stressors and SB-related family stress predicted a lower intercept for paternal marital satisfaction. Family support and family stress attributed to SB moderated the relationship between child vulnerability and maternal marital satisfaction. Observed family cohesion and child psychosocial quality of life moderated the relationship between family stressors and paternal marital satisfaction. CONCLUSION: Findings demonstrate the usefulness of the double ABCX model for this population.

Working memory and cognitive flexibility predict growth trajectories of sluggish cognitive tempo in youth with spina bifida.

As sluggish cognitive tempo (SCT) shows similar inattention and neuropsychological dysfunction as youth with spina bifida (SB), it is important to examine whether neuropsychological functioning may affect the development of SCT in this population. Participants were 140 youth with SB and their parents who participated in five waves of a longitudinal study across eight years (ages 8-15 years at Time 1). At Time 1, teacher-, mother-, and father-report of SCT showed 9%, 8.3%, and 5.3% impairment in SCT respectively compared to other youth in the sample. Growth curves were used to examine changes over time in mother-, father-, and teacher-reported SCT. Four neurocognitive variables were included (attention, working memory, cognitive flexibility, plan/organize) as predictor variables of SCT growth and intercepts. Mother, father, and teacher-report of SCT were included in separate models. Age and shunt status were included as covariates in the growth models. Inattentive symptoms were also included as a covariate in working memory, shift, and plan/organize models. Worse working memory at Time 1 predicted an increase in mother-reported SCT symptoms over eight years (ß = -.28) and poorer cognitive shifting skills predicted higher rates of teacher-reported SCT over time (ß = -.61). All other neurocognitive variables predicted the intercept, but not the slope of SCT symptoms. Results indicate that worse neurocognitive functioning is associated with higher levels of SCT symptoms and that worse working memory and cognitive flexibility at baseline predicted an increase in SCT symptoms as youth with SB age. It may be important for clinicians to monitor and assess levels of SCT in youth with SB.