Research Approaches to Improve the Care and Outcomes of People Living with Spina Bifida

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PROJECT/SUMMARY ABSTRACT Spina bifida (SB) is the most common permanently disabling birth defect. In the United States alone, it is estimated that more than 150,000 individuals live with SB. Given the high medical complexity of SB care many individuals with SB often exhibit numerous comorbidities (e.g., tethered cord syndrome, bowel/bladder incontinence, skin ulcers, cognitive deficits, etc.). Subsequently as adults, they often have a lower health-related quality of life, participate less in higher education, and have lower rates of employment. Much of this under- attainment can be traced back to early neurodevelopmental deficits. However, the early risk factors contributing to these poor neurodevelopmental outcomes are still insufficiently understood. Concurrently, with the proliferation of prenatal myelomeningocele repair and its associated risk for premature birth, the need for elucidating the impact of fetal repair on long-term neurodevelopmental outcomes has become more urgent than ever. Our early studies comparing minimally invasive fetal (fetoscopic) surgery to open-hysterotomy prenatal myelomeningocele repair, show that in addition to vaginal term births, those who were repaired fetoscopically demonstrate comparable neurosurgical, motor, and urological outcomes to those repaired using the open- hysterotomy approach. Furthermore, National Spina Bifida Patient Registry (NSBPR) publications have demonstrated that some racial/ethnic health disparities may exist in this national sample. The goal of this proposal is to investigate the outcomes related to prenatal myelomeningocele repair and concurrently investigate if differences in the receipt of subsequent care exist based on race/ethnicity. In Aim 1, we will examine the relationship between different approaches to fetal myelomeningocele repair and neurosurgical outcomes in order to identify best practice standards of care. In Aim 2, we will examine the relationship between neurogenic bladder and self-management, race/ethnicity, and fetal myelomeningocele repair in order to promote best practice guidelines for bladder continence. Lastly in Aim 3, we will evaluate for disparities in the relationship between race/ethnicity and the receipt of intervention for skin breakdown prevention or tethered cord syndrome in order to identify and address any difference identified. We expect that these studies will compare the neurosurgical and urologic outcomes of fetal MMC repair approaches in a national sample, and examine if disparities exist in relation to the receipt of interventions and outcomes.