Establishing the first community-centered Spina Bifida research agenda

Introduction: Spina bifida (SB) is the most common permanently disabling congenital defect. In 2016, the Spina Bifida Association (SBA) began updating the Guidelines for the Care of People with Spina Bifida (Guidelines). These Guidelines identified over 250 research gaps for the care of individuals with SB. The community of people living with SB prioritized these research gaps to create a formal research agenda that the SBA could support.

Objective: To present the process and the final research agenda created by individuals with SB, their care partners, clinicians, and researchers.

Design: A quantitative survey was developed to allow adults with SB and caregivers of those with SB to rank the impact of each of the 27 topical areas of the Guidelines. The survey was sent via SBA's database to English and Spanish-literate individuals. 1607 responses were captured and analyzed. Two focus groups were convened after survey analysis: adults with SB and caregivers of children with SB. Discussion outlines for the focus groups were developed based on the results from the survey and were used for known-group validation of the highest-ranked topics. The SBA then solicited caregivers of those with SB, adults with SB, and clinical communities to join its initial Research Advisory Council (RAC). Each group generated a list of top research questions to address the gaps in these highest-ranked impact areas. The SBA led discussion groups for each topic area to rank the proposed questions in order of importance to the SB community. They provided content validity and revisions to the language to improve interpretation by the SB community. The final SB Research Agenda was created from the final four to six ranked questions in each of the six topics.

Results: A ranking of findings from the quantitative survey identified the two most common topics impacting adults with SB and caregivers of both children and adults with SB were bowel incontinence and urinary incontinence.

Conclusion: A Research Agenda for SB was rigorously created to prioritize topic areas of highest impact as ranked by individuals in the SB community to fill the research gaps identified in the Guidelines. Bowel and urinary incontinence, both often treated by urologists, were ranked at the top. This agenda will be used to prioritize research efforts to improve the health of those with SB.