incontinence and quality of life in children with spina bifida

Conditions: Spina Bifida; Myelomeningocele

Summary: this study aims to develop an innovative, interactive tool for joint use by spina bifida patients and their urologists to identify patients interested in addressing their urinary and fecal incontinence and establish continence goal(s) they would like to achieve. to date, no such tool exists for use by spina bifida patients or urologists. this represents a major paradigm shift in the urologic care of pediatric sb patients. it will give children and families a voice in setting their personal goals for urinary and fecal incontinence, rather than relying on physicians' traditional clinical targets (e.g., absence of urinary incontinence, 4-hour dry interval). these traditional views fail to reflect the full patient experience of their ailment by underestimating symptoms and prioritizing only the most severe. this study represents the first time that such a process will be formalized before initiating urological therapy in children with sb (aim 2). additionally, this tool may help rule out interventions with a low chance of achieving desired goals and allow for a recalibration of unrealistic goals. the app will be useful for any child, regardless of urinary and fecal incontinence treatment history or underlying bladder pathology, as it will help describe personalized clinical treatment goals based on urinary incontinence, a characteristic all these patients share. this study will also capture the health-related quality of life (hrqol) impact of urinary and fecal incontinence improvement regardless of the treatment, whether behavioral, medical or surgical, many of which are available to patients regardless of age. a systematic therapeutic goal-setting tool will help in bringing precision medicine to the sb population.

phase 1: stakeholder group. the investigators will engage 30 dyads of children and adolescents with spina bifida aged 8-17 years old, who experienced any urinary and fecal incontinence in the past 4 weeks, and their caregivers in the exploration phase. these children and adolescents will be identified from among spina bifida patients receiving care from pediatric urology providers at riley hospital for children, indiana university health. as part of the exploration process, it is important to include patients that reflect the varied life experiences of those impacted by spina bifida. therefore, the investigators will aim to include children and adolescents with a range of physical and cognitive developmental delays. groups will be separated by gender to facilitate discussion and elicit any gender-specific urinary incontinence goals. the patient engagement core will create a smartphone-based "day in the life" diary toolkit that will be provided to the stakeholder group. the stakeholders will be asked to self-report their experiences and perspectives as they relate to being a child or adolescent with spina bifida and urinary and fecal incontinence, or taking care of someone with spina bifida with urinary incontinence. stakeholders will be asked to respond using video to various prompts such as documenting a typical day in their life, sharing their experiences. after completing the requested tasks, stakeholders will return the toolkit to the patient engagement. these self-reporting tools will be designed to be as usable as possible by as many participants as possible, taking into account common barriers caused by this condition, and relying on caregivers, as needed, to administer the tools. investigators anticipate that the kit will take approximately 3 hours to complete. through analysis and translation of the diaries and video, the patient engagement core will identify insights into patient-centered ui goals and areas of opportunity for design solutions. these identified opportunity spaces will inform the work for phase two, the creation phase. phase 2: in order to achieve a holistic and sustainable intervention, it is crucial that the investigators work with an interdisciplinary team that include designers, researchers, patients, family members, other caregivers, and health care providers in the process of creating the intervention. therefore, the investigators will invite 8-10 of the dyads from phase one to participate as members of the co-design panel which will be brought together to participate in a 2-4 hour co-design session. during this session, designers will share the insights gained from phase one work. panel participants will be asked to help refine these insights to define domains of patient-centered urinary incontinence goals. from there, the co-design panel will work to generate solutions for a tool to facilitate goal-setting and follow-up related to these domains (i.e., the tool, or app). this will be done through a series of generative, prototyping exercises with participants. for example, the group could work in small teams to generate ideas for their ideal goal-setting tool and then use sketches, storyboards, building materials like foam core, or verbal story-telling to prototype these ideas. the options for prototyping will be diverse to accommodate a spectrum of physical and cognitive abilities. these prototypes can then be used to generate discussion among the co-design panel, which will further refine the ideal aspects of the app from the perspective of children and adolescents with sb and their caregivers. the physician panel will then be invited to respond to a prototype of the app and give their input on how it can be improved to best fit into clinic workflow, address physician preferences, and work for a variety of patient-physician scenarios. these responses will be gathered through structured in-person interviews. phase 3: the investigators will ask patient/caregiver dyads with spina bifida (n=8), who were not part of the co-design panel in phase two, to utilize the tool in conjunction with their pediatric urologist during a pediatric urology clinic visit at riley hospital. the investigators will comply with all irb requirements at the study institution for the inclusion of patients in research, including obtaining the necessary consents/assents. testing process. following the clinic visit, the patient/caregiver will participate in cognitive interviews. they will be asked to describe their impressions of the prototype app through the use of scripted verbal probes ("what do you think is being represented here?" "describe what you would do next.") and spontaneous probes ("what about this picture/sentence/etc., makes you feel that way?"), the investigators will determine if the prototype app is functioning as expected, or any key elements were missed. the investigators will also ask for feedback from the urologists on how well the tool functioned within the clinic setting to help in this iterative validation process. this will be accomplished by conducting interviews with approximately 6 physicians. phase 4: the investigators will recruit children and adolescents with sb (8-17 years old) who are followed at riley hospital pediatric urology outpatient clinic and their primary caregiver. the primary caregiver must be the patient's legal guardian. well over 200 children and adolescents with sb are seen at this facility annually. each week, a research assistant (ra) will be given a list of sb patients within the desired age range who have an upcoming appointment in two settings: the pediatric urology clinic or the multidisciplinary spina bifida clinic. the ra will approach the patient and their primary caregiver upon arrival at the clinic appointment to gauge their interest in study participation. if the dyad indicates potential interest, the ra will take them to a private area to further discuss the details of the study, determine eligibility, and then obtain consent from the legal caregiver and assent from the patient wishing to enroll in the study. over a 12-month period, the investigators will enroll 92 children and adolescents with sb and their primary caregivers. after enrollment in the study, the ra will administer the app, and collect baseline data regarding self-reported urinary and fecal incontinence and hrqol. the patient and their caregiver will then meet with their urologist for the scheduled clinic appointment. after being enrolled at baseline, participants will continue with standard care for 12 months (while completing ui, hrqol, and other questionnaires), at which point the app is introduced and the goal is set, and then they are followed for another 12 months. the urologist will be provided with the completed app and will utilize this tool to verify/discuss the goal selected, and then discuss plans for treatment in order to achieve this goal. goal attainment, ui, and hrqol will be assessed at 6 and 12 months after the app is introduced. children with sb are typically seen in the outpatient clinic every 6-12 months. therefore, these follow-up assessments will occur in-person at a clinic appointment, or, if not possible, investigators will contact the participant by telephone to notify them that an email link to a secure website to complete the necessary assessments will be sent. to minimize loss to follow-up, investigators will ask for 3 different phone numbers with at least one being a landline (if available). investigators will also ask for two email addresses. up to 3 attempts will be made to reach participants.

Eligibility criteria:
inclusion criteria: * spina bifida * 8-17 years old * followed at riley hospital pediatric urology outpatient clinic * primary caregiver is patient's legal guardian * urinary incontinence in the past 4 weeks * fecal incontinence in the past 4 weeks * normal to mildly impaired cognitive development * english language literacy * intent to receive care at the riley pediatric urology clinic for th extent of the study the physician panel will be composed of pediatric urologists. exclusion criteria: * children with significant cognitive impairment, as they will not be able to express their urinary and fecal incontinence goals. * patients who underwent a genitourinary procedure or a bowel procedure in the past 4 weeks, since these patients are often temporarily catheterized and therefore not experiencing their typical level of urinary and fecal incontinence.