A systematic review and meta-analysis of pediatric integrated primary care for the prevention and treatment of physical and behavioral health conditions.

OBJECTIVE: To evaluate the effects of behavioral health interventions delivered within pediatric integrated primary care models on clinical outcomes. METHODS: We searched Medline, EMBASE, CENTRAL, PsycINFO, and SCOPUS for studies published from January 1, 1998, to September 20, 2023. We included studies that evaluated onsite behavioral health integration in pediatric primary care using a comparator condition (usual, enhanced usual care, or waitlist). Outcome data on symptom change, impairment/quality of life, health indicator, and behavior change were extracted using Covidence software. The Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA) guideline was followed Risk of bias analysis was conducted using the Cochrane Risk of Bias tool. We used multilevel meta-analysis to synthesize multiple outcomes nested within studies. Open Science Foundation pre-registration: #10.17605/OSF.IO/WV7XP. RESULTS: In total, 33 papers representing 27 studies involving 6,879 children and caregivers were included. Twenty-four studies were randomized controlled trials and three were quasi-experimental designs. Seventeen papers reported on treatment trials and 16 reported on prevention trials. We found a small overall effect size (SMD = 0.19, 95% confidence interval [0.11, 0.27]) supporting the superiority of integrated primary care to usual or enhanced usual care. Moderator analyses suggested similar effectiveness between co-located and integrated models and no statistically significant differences were found between treatment and prevention trials. CONCLUSIONS: Results suggest that integrated primary care is superior to usual and enhanced usual care at improving behavior, quality of life, and symptoms. Integrated primary care research needs improved standards for reporting to promote better synthesis and understanding of the literature.

Cecostomy tubes improve bowel continence for pediatric patients with spina bifida: A retrospective analysis of outcomes from a single clinic.

PURPOSE: Pediatric patients with spina bifida often experience neurogenic bowel dysfunction. Although cecostomy tubes could improve bowel continence, their effectiveness is not well established in this population. The aims of this study were to better understand the effectiveness of cecostomy tubes relative to other management strategies (between-subject) and to explore their effectiveness among patients who received these placements (within-subject). METHODS: Retrospective analysis of data from pediatric patients enrolled in a national spina bifida patient registry (n = 297) at a single multidisciplinary clinic was performed, covering visits between January 2014 -December 2021. Linear and ordinal mixed effect models (fixed and random effects) tested the influence of cecostomy status (no placement vs placement) and time (visits) on bowel continence while controlling for demographic and condition-specific covariates. RESULTS: Patients with cecostomy tubes had higher bowel continence compared to patients without placements (B = 0.695, 95% CI [0.333, 1.050]; AOR = 2.043, p = .007). Patients with cecostomy tubes had higher bowel continence after their placements compared to before (B = 0.834, 95% CI [0.142, 1.540]; AOR = 3.259, p = 0.011). CONCLUSION: Results indicate cecostomy tubes are effective for improving bowel continence in this pediatric population. Future research is needed to conduct risk analyses and determine the clinical significance of these effects.

Association between age of starting clean intermittent catheterization and current urinary continence in individuals with myelomeningocele.

INTRODUCTION: Patients with myelomeningocele often use clean intermittent catheterization (CIC) for renal preservation and to promote urinary continence. While starting CIC at an early age is associated with better renal outcomes, the impact of age of CIC initiation on continence outcomes has not been examined. OBJECTIVE: To examine whether earlier CIC initiation is associated with higher likelihood of current urinary continence for patients with myelomeningocele. STUDY DESIGN: Data of patients aged ≥5 years at last visit were obtained from 35 spina bifida clinics participating in the National Spina Bifida Patient Registry from 2013 to 2018. Sociodemographic characteristics, disease characteristics, and current bladder management strategies were collected. Via univariate and multiple logistic regression models, the latter conducted controlling for all variables associated with current continent status, associations between continence and sociodemographic factors, condition characteristics, and age CIC began (<3 years of age, 3-5 years, 6-11 years, ≥12 years) were analyzed. RESULTS: Data from 3510 individuals were included (mean age at last visit = 17.0 years, range 5.0-88.7). The sample was evenly distributed by sex (52% female); most individuals were non-Hispanic White (62.6%). The majority of patients (55.2%) started CIC before age 3 years. Continence varied markedly across those who never started CIC (0.6% of patients were continent) and those who started at any age (range 35.3-38.5%). Among those who started CIC, the magnitude of the association was not proportional to age CIC was started. Compared with those who started CIC at age 12 or older, estimated adjusted odds ratio of being continent ranged from 1.04 (6-11 years, 95% CI, 0.72-1.52) to 1.25 (<3 years, 95% CI, 0.89-1.76). DISCUSSION: Although CIC may be positively associated with achieving urinary continence in individuals with myelomeningocele, we could not demonstrate that younger age at CIC initiation increased the likelihood of achieving this goal. Limitations include lack of data on reason for starting CIC, urodynamic data, and the observational nature of data collection. CONCLUSIONS: Further study is needed addressing limitations of the current investigation to determine if urinary continence outcomes are influenced by the age of starting CIC among patients with myelomeningocele.

Differences in continence rates in individuals with spina bifida based on ethnicity.

BACKGROUND: Ethnic disparities in continence rates in spina bifida (SB) have been studied regionally but not nationally. National SB Patient Registry (NSBPR) data were analyzed to explore differences in prevalence of bowel and bladder continence and interventions between Hispanics/Latinos and others. METHODS: Participants 5 to 21 years were categorized into Hispanic/Latino and non-Hispanic/non-Latino. Bladder/bowel continence was defined as dry/no involuntary stool leakage during the day or none/⩽ monthly incontinence. Chi-square test, Wilcoxon Two Sample Test, and generalized estimating equation (GEE) were used for statistical analysis. RESULTS: Twenty-five percent of the 4,364 patients were Hispanic/Latino. At their most recent clinic visit, Hispanics/Latinos demonstrated lower rates of urinary continence (38.6% vs. 44.9%; p= 0.0003), bowel continence (43.9% vs. 55.8%, p< 0.0001), private insurance (p< 0.0001), bowel (p< 0.0001) or bladder surgeries (p= 0.0054), and more vesicostomies (p= 0.0427) compared to others. In multiple GEE models, Hispanic/Latino participants demonstrated lower odds of bowel continence as compared to non-Hispanic/non-Latino participants (estimated odds ratio, 0.82, 95% CI, 0.72-0.94, p= 0.0032). CONCLUSIONS: After controlling for covariates, Hispanics/Latinos with SB are less likely to report bowel continence. Clinicians are encouraged to consider the risk of negative health disparities for Hispanic patients with SB and work to mitigate this risk.

Impacting Pediatric Primary Care: Opportunities and Challenges for Behavioral Research in a Shifting Healthcare Landscape.

Behavior analysts have long recognized the potential of a partnership with pediatric medicine as an opportunity to expand the influence of behavior analysis and positively impact population health. Despite significant achievements in this domain, the impact of behavioral science on the daily practice of pediatrics has been limited. In this commentary, the authors argue that the current health care and research environments are ripe for a renewed focus on behavioral modification in pediatric primary care, with a particular emphasis on the study of high-frequency, low-intensity problems. They provide some analysis of why behavioral pediatrics has failed to gain traction in primary care, describe aspects of the current primary care practice and research landscapes that provide opportunities for an expanded portfolio of research, identify several exemplars from the behavior analytic literature that have influenced pediatric primary care or have the potential to do so, and make recommendations for producing influential data.

Longitudinal Study of Bladder Continence in Patients with Spina Bifida in the National Spina Bifida Patient Registry.

PURPOSE: Achieving bladder continence in individuals with spina bifida is a lifetime management goal. We investigated bladder continence status through time and factors associated with this status in patients with spina bifida. MATERIALS AND METHODS: We used National Spina Bifida Patient Registry data collected from 2009 through 2015 and applied generalized estimating equation models to analyze factors associated with bladder continence status. RESULTS: This analysis included 5,250 participants with spina bifida in a large, multi-institutional patient registry who accounted for 12,740 annual clinic visit records during the study period. At last followup mean age was 16.6 years, 22.4% of participants had undergone bladder continence surgery, 92.6% used some form of bladder management and 45.8% reported bladder continence. In a multivariable regression model the likelihood of bladder continence was significantly greater in those who were older, were female, were nonHispanic white, had a nonmyelomeningocele diagnosis, had a lower level of lesion, had a higher mobility level and had private insurance. Continence surgery history and current management were also associated with continence independent of all other factors (adjusted OR and 95% CI 1.9, 1.7-2.1 and 3.8, 3.2-4.6, respectively). The association between bladder management and continence was stronger for those with a myelomeningocele diagnosis (adjusted OR 4.6) than with nonmyelomeningocele (adjusted OR 2.8). CONCLUSIONS: In addition to demographic, social and clinical factors, surgical intervention and bladder management are significantly and independently associated with bladder continence status in individuals with spina bifida. The association between bladder management and continence is stronger in those with myelomeningocele.

Variation in bowel and bladder continence across US spina bifida programs: A descriptive study.

PURPOSE: Continence is low in individuals with spina bifida, but published prevalence varies markedly across studies. The objective of this study was to examine bladder and bowel continence among patients served by multidisciplinary clinics participating in the National Spina Bifida Patient Registry and to examine whether variation in prevalence exists across clinics. METHODS: Data were obtained from patients 5 years and older from March 2009 to December 2012. Data were gathered at clinic visits using standardized definitions. RESULTS: Data from 3252 individuals were included. Only 40.8% of participants were continent of urine; 43% were continent of stool. Bladder and bowel continence differed by spina bifida type, with those with myelomeningocele having significantly lower reported prevalence of continence than those with other forms of spina bifida. Bladder and bowel continence varied across registry sites. Adjustment based on demographic and condition-specific variables did not make substantive differences in prevalence observed. CONCLUSION: Less than half of spina bifida patients served in multidisciplinary clinics report bladder or bowel continence. Variability in prevalence was observed across clinics. Further research is needed to examine if clinic-specific variables (e.g., types of providers, types of interventions used) account for the observed variation.

Normal IQ is possible in Smith-Lemli-Opitz syndrome.

Children with Smith-Lemli-Opitz syndrome (SLOS) are typically reported to have moderate to severe intellectual disability. This study aims to determine whether normal cognitive function is possible in this population and to describe clinical, biochemical and molecular characteristics of children with SLOS and normal intelligent quotient (IQ). The study included children with SLOS who underwent cognitive testing in four centers. All children with at least one IQ composite score above 80 were included in the study. Six girls, three boys with SLOS were found to have normal or low-normal IQ in a cohort of 145 children with SLOS. Major/multiple organ anomalies and low serum cholesterol levels were uncommon. No correlation with IQ and genotype was evident and no specific developmental profile were observed. Thus, normal or low-normal cognitive function is possible in SLOS. Further studies are needed to elucidate factors contributing to normal or low-normal cognitive function in children with SLOS.

A Survey of Parents' Perceptions and Use of Time-out Compared to Empirical Evidence.

OBJECTIVE: To assess parents' perceptions and use of time-out (TO) in contrast to empirical indications and examine the relationship between reported implementation procedures and perceived effectiveness. METHODS: We surveyed parents of preschool and school-age children (n = 401, aged 15 months to 10 years) at well-child visits with regard to their awareness, perception, and usage of TO. Parents were specifically surveyed regarding TO components that have been empirically evaluated or pertain directly to its underlying behavioral principles. Descriptive analyses, group comparisons, and correlational analyses were used to characterize responses and evaluate the relationship between TO administration variation and perceived effectiveness. RESULTS: Most parents (76.8%) reported using TO in response to misbehavior, but a large majority of these parents (84.9%) reported implementing TO in a manner counter to empirical evidence. Parents who endorsed TO as effective varied significantly from those who did not on key implementation components (eg, use of a single warning). Further, several reported implementation practices were correlated with perceived effectiveness and challenging child behavior. For example, requiring a child to be calm before ending TO was positively correlated with perceived effectiveness. CONCLUSIONS: These results cement TO as a widely disseminated practice but cast doubt on the fidelity with which it is typically implemented. Better methods of educating parents on evidence-based discipline are needed.

A Pilot Study of the Association of Markers of Cholesterol Synthesis with Disturbed Sleep in Smith-Lemli-Opitz Syndrome.

OBJECTIVE: Smith-Lemli-Opitz syndrome (SLOS) is a rare genetic disorder characterized by cholesterol synthesis impairment. A host of physical, developmental, and behavioral presentations are associated with SLOS, many of which have been related with disorder severity. Sleep disturbance is commonly reported in SLOS. This study is the first to examine the association between sleep disturbance and biomarkers of cholesterol synthesis defect. METHOD: Twenty youth with SLOS participated. Biomarkers of cholesterol synthesis were obtained, including plasma sterols (i.e., 7-dehydrocholesterol, 8-dehydrocholesterol, and cholesterol), mevalonic acid, and 24-S hydroxycholsterol. A ratio of plasma cholesterol precursors to cholesterol levels was used as a measure of biochemical severity. Parents reported their children's sleep problems using the Children's Sleep Habits Questionnaire. RESULTS: Most markers of cholesterol synthesis disruption were associated with overall sleep disturbance. Biochemical severity of SLOS was also associated with specific sleep problems (e.g., decreased sleep duration and increased sleep onset delay) and was identified as a significant predictor of these factors. CONCLUSION: This study is the first to demonstrate associative relationships between cholesterol levels and sleep disturbance in youth with SLOS. These results add to the current understanding of how cholesterol levels may contribute to the behavioral phenotype of SLOS. These findings may inform future studies related to the role cholesterol synthesis defects play in the behavioral phenotype of SLOS and, subsequently, modalities of intervention for behavioral symptoms.

Videoconferencing for Teens With Diabetes: Family Matters.

BACKGROUND: Adolescence is a developmental period associated with increased difficulty managing diabetes. During adolescence family functioning, including miscarried helping, family conflict, and acceptance of illness, is an important predictor of adherence to treatment recommendations. Multiple barriers exist to receiving behavioral health interventions to address suboptimal adherence. We hypothesized that behavioral family systems therapy-diabetes (BFST-D) delivered via telehealth would yield changes in family functioning that were not significantly different than changes in clinic-based treatment. Furthermore, that BFST-D would significantly improve overall family functioning. METHODS: Ninety adolescent participants and their parents were randomized to receive BFST-D via telehealth or traditional (Clinic) treatment conditions. Repeated measures ANOVAs were used to assess changes in mean scores across pre, post, and follow-up assessments. Mediation analyses were conducted using methods outlined by Sobel and were confirmed by bootstrapping. RESULTS: Changes in miscarried helping, family conflict and adjustment to illness were not significantly different across groups. Overall, clinically significant improvements were identified in youth- and parent-reported miscarried helping, family conflict, and acceptance of illness. Reductions in family conflict mediated the relationship between changes in miscarried helping and acceptance of illness. In addition, improvements in family functioning were associated with changes in adherence and glycemic control. CONCLUSIONS: Results provide strong support for BFST-D (and similar interventions) delivered via telehealth as yielding outcomes no different than clinic-based treatment. In addition, further support was provided for the effectiveness of BFST-D.

The effectiveness of bowel and bladder interventions in children with spina bifida.

AIM: Using the World Health Organization International Classification of Functioning, Disability and Health (ICF), the aim of this study was to identify effective strategies for managing urinary and bowel complications resulting from spina bifida. METHOD: Charts of 210 children between 4- and 13-years-old with spina bifida were reviewed to quantify medical interventions and continence status. Standardized quality of life (QOL) questionnaires were administered to a subset of participants; child and parent interviews were carried out to examine the experience of living with bowel and bladder incontinence. Practitioners were also interviewed to understand their perspectives of intervention effectiveness. RESULTS: Chart review indicated less than half of children were continent for bowel and bladder. More variability existed in bowel continence programs, and practitioners considered bowel continence more difficult to achieve than bladder continence. No significant associations were found between continence status and QOL measures. Interviews, however, reflected how managing continence at home and school more broadly affects QOL. Among practitioners, some focused primarily on optimizing physical health while others focused on activity and participation. INTERPRETATION: While continence is a goal, programs used to achieve this are individualized and outcomes may be affected by differential treatment effects, environmental factors, and/or stigma experienced by children.

Dissemination of Evidence-Based Behavioral Advice via Video in Pediatric Primary Care: An Acceptance and Utilization Study.

BACKGROUND: Research suggests that multimedia-based interventions possess advantages for disseminating safe and effective methods of behavior management to parents in pediatric primary care; however, little is known about their utilization in real-life settings. In order to maximize the impact of multimedia resources, more knowledge regarding dissemination and implementation is needed. OBJECTIVE: To examine provider and parental perception and utilization of videos designed to communicate evidence-based parenting strategies for disruptive behavior. Videos were available in clinic and online. RESULTS: Both provider and parent perceptions of the videos were largely positive. However, of 240 parents surveyed, only 33% were aware of the availability of videos subsequent to a well-child visit. Parents were unlikely to view the videos if they did not do so as part of their child's health care visit. CONCLUSION: Multimedia interventions for behavior management are likely to be well received, but systematic methods of implementation are needed. Further study of dissemination of multimedia interventions is merited.

Seeing Is Believing: Using Skype to Improve Diabetes Outcomes in Youth.

OBJECTIVE: The objective of this study was to compare the relative effectiveness of two modes of delivering Behavioral Family Systems Therapy for Diabetes (BFST-D) to improve adherence and glycemic control among adolescents with type 1 diabetes with suboptimal glycemic control (HbA(1c) ≥9.0% [≥74.9 mmol/mol]): face to face in clinic (Clinic) and Internet videoconferencing (Skype) conditions. RESEARCH DESIGN AND METHODS: Adolescents aged 12 to 18 years and at least one adult caregiver were randomized to receive BFST-D via the Clinic or Skype condition. Participants completed up to 10 therapy sessions within a 12-week period. Changes in youth- and parent-reported adherence and glycemic control were compared before and after the intervention and at follow-up assessment. RESULTS: Using an intent-to-treat analytic approach, no significant between-group differences were identified between the before, after, and follow-up assessments. Groups were collapsed to examine the overall effects of BFST-D on adherence and glycemic control. Results identified that statistically significant improvements in adherence and glycemic control occurred from before to after the intervention; improvements were maintained at 3-month follow-up. CONCLUSIONS: Delivery of BFST-D via Internet-based videoconferencing is viable for addressing nonadherence and suboptimal glycemic control in adolescents with type 1 diabetes, potentially reducing important barriers to care for youth and families.

Depressive Symptoms in a Trial Behavioral Family Systems Therapy for Diabetes: A Post Hoc Analysis of Change.

OBJECTIVE: The objective was to test whether Behavioral Family Systems Therapy for Diabetes (BFST-D), an evidence-based family therapy, produces individual changes in depressive symptoms for adolescents with type 1 diabetes in suboptimal glycemic control (HbA(1c) ≥9.0% [≥74.9 mmol/mol]). RESEARCH DESIGN AND METHODS: Data were from a randomized controlled trial (RCT) comparing two modes of BFST-D delivery: in clinic versus Internet videoconferencing. There were no significant differences between groups in the RCT, so groups were collapsed into a within-group prepost design for secondary analyses. A multiple regression analysis was performed to test for mediation of treatment outcomes by changes in family processes. RESULTS: Significant improvements in glycemic control, depressive symptoms, and family functioning were found from pre- to posttreatment. A multiple regression analysis for within-subject mediation indicated that improvements in depressive symptoms were partially mediated by improvements in parent-youth conflict; however, family process changes did not mediate diabetes health outcomes. CONCLUSIONS: In addition to improving treatment adherence and glycemic control, BFST-D has collateral benefits on depressive symptoms.

Systematic review and meta-analysis of behavioral interventions for fecal incontinence with constipation.

BACKGROUND: Multiple treatments exist for fecal incontinence. However, the relative and additive influence of commonly used behavioral approaches remains unclear. OBJECTIVE: We conducted a systematic review of randomized controlled trials to synthesize the effects of behavioral treatment of fecal incontinence with constipation in children aged 4-18 years. Mixed treatment comparisons (MTCs) and random effects models were used to analyze outcomes. Risk of bias and quality of evidence were rated. RESULTS: Although 10 studies were identified for MTCs, results did not yield reliable or valid estimates. Four studies were retained for random effects pooled outcome analysis. Results indicated that behavioral intervention was more effective than control conditions for author-defined success and soiling frequency. CONCLUSION: Although evidence supports behavioral treatments for fecal incontinence with constipation in children, available evidence is limited. More and higher-quality trials are needed to better understand the relative effects of different treatments, including behavioral strategies.

Power of magic hands: parent-driven application of habit reversal to treat complex stereotypy in a 3-year-old.

OBJECTIVE: The authors assessed the effectiveness of habit reversal training (HRT) to treat a complex motor stereotypy in a healthy 3-year-old female. METHOD: This data-based case study involved training parents in HRT to deliver the parent-driven intervention to the child. The frequency of the child's behaviors was estimated daily in 30-min intervals by her parents. RESULTS: Outcomes supported the effectiveness of the intervention, with the estimated frequency of the stereotypy decreasing from occurring during approximately 85% of recorded intervals to less than 2% over a period of 4 weeks. Further record keeping over 19 weeks suggested treatment gains were generally maintained over time. CONCLUSIONS: The current case study provides preliminary evidence supporting the effectiveness of modified HRT to reduce stereotypies in young children. Further, data suggest that the intervention may be extended to younger ages by teaching parents how to facilitate treatment delivery.

Behavioral health care for adolescents with poorly controlled diabetes via Skype: does working alliance remain intact?

BACKGROUND: Increasingly various technologies are being tested to deliver behavioral health care. Delivering services via videoconferencing shows promise. Given that the patient-provider relationship is a strong predictor of patient adherence to medical regimens, addressing relationship quality when services are not delivered face-to-face is critical. To that end, we compared the therapeutic alliance when behavioral health care was delivered to youth with poorly controlled type 1 diabetes mellitus (T1DM) and their caregivers in-clinic with the same services delivered via Internet-based videoconferencing (i.e., Skype™). METHODS: Seventy-one adolescents with poorly controlled T1DM (hemoglobin A1c ≥9%) and one of their caregivers received up to 10 sessions of a family-based behavioral health intervention previously shown to improve adherence to diabetes regimens and family functioning; 32 were randomized to the Skype condition. Youth and caregivers completed the working alliance inventory (WAI), a 36-item measure of therapeutic alliance, at the end of treatment. Additionally, the number of behavioral health sessions completed was tracked. RESULTS: No significant differences in WAI scores were found for those receiving behavioral health care via Skype versus in-clinic. Youth WAI goal and total scores were significantly associated with the number of sessions completed for those in the clinic group. CONCLUSION: Behavioral health can be delivered to youth with T1DM via Internet-based videoconferencing without significantly impacting the therapeutic relationship. Thus, for those adolescents with T1DM who require specialized behavioral health care that targets T1DM management, Internet-based teleconferencing represents a viable alternative to clinic-based care.

Challenging behavior in Smith-Lemli-Opitz syndrome: initial test of biobehavioral influences.

OBJECTIVE: To study challenging behavior (destruction, aggression, self-injury, stereotypy) in children with Smith-Lemli-Opitz syndrome (SLOS) using a biobehavioral model that helps distinguish biological from socially mediated variables influencing the behavior. BACKGROUND: SLOS is an autosomal-recessive syndrome of multiple malformations and intellectual disability resulting from a genetic error in cholesterol synthesis in all cells and tissues, including brain. The exact cause of the challenging behavior in SLOS is unclear, but defective brain cholesterol synthesis may contribute. Because the precise genetic and biochemical etiology of SLOS is known, this disorder is a good model for studying biological causes of challenging behavior. METHOD: In a preliminary application of a biobehavioral model, we studied the association between cholesterol levels (as a biochemical indicator of disease severity) and behavior subtype ("biological" vs "learned") in 13 children with SLOS. Parents completed a questionnaire that categorized challenging behavior as influenced primarily by social or nonsocial (thus, presumably biological) factors. RESULTS: The severity of the cholesterol synthesis defect correlated significantly with behavior subtype classification for 1 of 2 challenging behaviors. Greater severity of the cholesterol synthesis defect was associated with behavior being classified as primarily influenced by biological factors. CONCLUSION: The interplay between challenging behavior and defective cholesterol synthesis in SLOS may help explain biological influences on the behavior. Our findings have implications for research on the effectiveness of behavioral and medical treatments for behavioral difficulties in SLOS and other neurodevelopmental disorders.

Testing the feasibility of a National Spina Bifida Patient Registry.

BACKGROUND: The purpose of this study was to describe the development and early implementation of a national spina bifida (SB) patient registry, the goal of which is to monitor the health status, clinical care, and outcomes of people with SB by collecting and analyzing patient data from comprehensive SB clinics. METHODS: Using a web-based, SB-specific electronic medical record, 10 SB clinics collected health-related information for patients diagnosed with myelomeningocele, lipomyelomeningocele, fatty filum, or meningocele. This information was compiled and de-identified for transmission to the Centers for Disease Control and Prevention (CDC) for quality control and analysis. RESULTS: A total of 2070 patients were enrolled from 2009 through 2011: 84.9% were younger than 18 years of age; 1095 were women; 64.2% were non-Hispanic white; 6.5% were non-Hispanic black or African American; and 24.2% were Hispanic or Latino. Myelomeningocele was the most common diagnosis (81.5%). CONCLUSIONS: The creation of a National Spina Bifida Patient Registry partnership between the CDC and SB clinics has been feasible. Through planned longitudinal data collection and the inclusion of additional clinics, the data generated by the registry will become more robust and representative of the population of patients attending SB clinics in the United States and will allow for the investigation of patient outcomes.