Abstract
PURPOSE:
In order to transition to adulthood and independence, youth with spina bifida must assume significant self-management responsibilities including monitoring for shunt malfunction, maintaining intact skin in areas that are insensate, and maintaining proper bowel and bladder function. Validated measures of specific spina bifida self-management skills are lacking and this hampers the ability of clinical personnel to support successful transition for youth with spina bifida.
METHODS:
We developed a self-report measure specific to SB self-management skills consistent with the framework of the Transition Readiness Assessment Questionnaire (TRAQ). To test the predictive validity of the tool we surveyed 90 youth and young adults ages 12-25 with spina bifida attending a multidisciplinary clinic participating in the National Spina Bifida Patient Registry (NSBPR).
RESULTS:
Adjusted for age, gender, race, insurance status and lesion level, higher scores on the TRAQ-SB (increased self-management) were negatively associated with urinary incontinence in the past month. Only lesion level, and not TRAQ-SB scores, was a significant predictor of stool incontinence and skin breakdown.
CONCLUSIONS:
Higher TRAQ-SB scores are negatively associated with bladder incontinence in youth with spina bifida. While stool continence and skin breakdown were not associated with TRAQ-SB scores, this relation is complex and may be obfuscated by either reporting bias or outcome measurement bias. To further refine the questionnaire and understand this relationship we need to field it prospectively in the SB network with larger samples. The TRAQ-SB questionnaire, however, does have value in the clinical setting to help promote the acquisition of specific self-management skills among youth with spina bifida.
Background
Health Care Transition (HCT), is defined as the process of the “purposeful, planned movement of adolescents and young adults with chronic physical and medical conditions from child-centered to adult-oriented health care systems,” [1]. The transition and eventual transfer from pediatric to adult healthcare can be challenging for an adolescent with a chronic condition [2]. As adolescents approach adulthood and assume greater autonomy, they need to gain the skills required to manage their own health and health care independently, (e.g. taking their medications reliably; making and keeping appointments; and communicating effectively with healthcare providers). This set of self-management skills constitutes “Transition Readiness.” A growing body of literature suggests that many adolescents with chronic conditions do not acquire transition readiness skills and, as a result, experience a decline in their adherence to prescribed medical regimens (e.g. taking medications on time and in the correct amount, attending scheduled follow-up visits with doctors) and suffer poor outcomes such as increased preventable morbidity and even mortality [3, 4, 5, 6].
In addition to the above skills, youth with spina bifida have significant demands for self-management specific to their condition including monitoring for shunt malfunction, maintaining intact skin in areas that are insensate, and maintaining proper bowel and bladder function [7]. Spina bifida is a complex condition with a wide range of presentation depending upon the level of the spinal lesion and the degree of denervation of the bladder, bowel and skin. Patients with spina bifida are at particularly high risk for poor outcomes during transition. Youth and young adults with spina bifida with low self-management scores were more likely to have complications compared to those with higher self-management scores [8]. Moreover, when compared to children and adolescents with spina bifida, the young adults with spina bifida experience more frequent preventable hospitalizations and emergency room visits [9].
Previously we developed a disease independent measure of self-management skills entitled the Transition Readiness Assessment Questionnaire (TRAQ) that measures self-management skills including managing medications, making appointments, tracking health issues, and communicating with providers. Research demonstrates that TRAQ scores increase with age and are sensitive to interventions by providers designed to increase transition readiness and self-management skills, such as goal setting and texting reminder [10, 11, 12].
To complement the general health care transition skills assessed in the TRAQ, we developed an 11-item supplement to the TRAQ to assess self-management skills specific for spina bifida, including questions that assess the self-management of urine and stool continence management, and skin and shunt maintenance. In a companion article in this issue, we demonstrate that this 11-item scale has good internal reliability (
Methods
Sample
Patients with spina bifida ages 12–25 who had attended for at least one year a multidisciplinary clinic participating in the National Spina Bifida Patient Registry (NSBPR) were asked to participate in the study and complete the TRAQ-SB and allow their NSBPR information to be abstracted. During late 2016 and early 2017, ninety-three consecutive patients of the appropriate age range were approached and consented to participate. Of the 93 patients approached, 90 agreed to participate and completed the TRAQ-SB.
Data collection
Systematic data were collected on their condition, demographics, bowel and bladder continence, and skin breakdown according to the definitions and protocol developed by the NSBPR (details on the NSBPR are available elsewhere [13]. The data from the electronic medical record and SB registry was abstracted and linked with participants’ responses to the TRAQ and TRAQ-SB.
Variables and measures
Demographic variables
Clinical demographic information was abstracted from the electronic medical record. Data collection techniques and variable definitions utilized the methodology of the National Spina Bifida Patient Registry (NSBPR) [14]. Specifically, we obtained age, sex, race, insurance status, individualized education plan status, lesion level, and type of spina bifida. Race included the following categories: black, white and other. Functional lesion level, which in its original form indicated either right or left and lesion level with ten categories, and we collapsed into five categories from highest/most severe impairment to lowest/mildest impairment: thoracic, high-lumbar, mid-lumbar, low-lumbar, and sacral. We recorded whether or not the participant had an Individual Educational Plan (IEP), as a proxy for presence of a learning disability. Insurance status was categorized as commercial, Medicaid, Medicare, Tri-Care, State High-Risk Plan, and other.
Transition Readiness Assessment Questionnaire-Spina Bifida supplement (TRAQ-SB)
The TRAQ-SB is a newly developed 11-item scale created to assess aspects of SB self-care and self-management including urine and stool incontinence, prevention of skin breakdown and awareness of signs of shunt malfunction (see Appendix for the full 20 item TRAQ and 11 item SB supplement). For more information on the development and psychometric testing of the TRAQ-SB, please see Johnson et al. [13]. For stool, urine and skin management we asked three questions on each area: 1. Do you practice management activities as recommended for…; 2. Do you know how to tell the warning signs of problems with…and, 3. Do you take the necessary steps to address a __ problem? An example statement from the scale includes, “Do you take steps to protect your skin from potential damage, pressure sores, or infection?”. Given the wide range of anatomic differences in bowel and bladder anatomy among persons with spina bifida and the heterogeneous nature of the self-management interventions required to maintain continence in this population, we structured the questions as general assessments of their behaviors and knowledge so that they would apply to all youth with spina bifida.
Youth and young adults were asked to rate their behavior, knowledge and action in response to problem on a scale of one to five based on the Stages of Change Theoretical Model [15], designed to measure youth’s stages of change from: 1
Clinical outcomes
Urinary incontinence
Urinary incontinence was assessed by dichotomizing the 6-point scale as defined by the NSBPR. Original responses ranged from 1 to 6, where 1 represents cannot assess, 2 represents greater than or equal to once per day (daily), 3 – less than once per day, more than or equal to once per week (weekly), 4 – less than once per week, greater than or equal to once per monthly (monthly), 5 – less than once per month (less than monthly), and 6 – representing never. For the purposes of this study, a response of “cannot assess,” was treated as missing. Urinary continence was defined as experiencing stool leakage less than once a month (answers 5 or 6 above) versus more than once a month (answers 2–4 above).
Stool incontinence
Stool incontinence was also assessed on a similar scale and we dichotomized the 6-point scale from the NSBPR in similar fashion. Therefore, stool continence was defined as experiencing stool leakage less than once a month (answers 5 or 6) versus more than once a month (answers 2–4).
Skin breakdown
Skin breakdown was assessed using a dichotomous yes/no response item to the question asking whether or not the youth had a skin breakdown in the last year.
Time relationship between self-management response and outcomes
At one visit per year, the data for clinical outcomes is entered into the National Spina Bifida Patient Registry. The questions regarding continence in the registry ask about the prior month (for skin breakdown the time frame is the past year). Patients are typically seen more than once per year. The TRAQ-SB questionnaire was filled out during visits during late 2016 and early 2017 the TRAQ-SB questions were asked at the same visit as the questions about skin breakdown and stool and urine incontinence. The TRAQ-SB questions asked about patients’ current (present tense) practice or knowledge (Do you practice management activities as recommended for…; Do you take the necessary steps to address a __ problem). Therefore, both the TRAQ-SB questions and the Spina Bifida Patient Registry questions ask about current state.
Analytic plan
All statistical analyses were conducted in IBM SPSS Statistics 24. Univariate statistics were used to describe the total sample, scale score means, standard deviations, and distributions. We used ANOVA to test unadjusted associations between our primary outcomes as well demographic variables and TRAQ-SB scores. Prior to conducting logistic regression, we examined the variables in the model for dependent variable structure, observation independence, absence of multicollinearity, linearity of independent variables and log odds, and found that the assumptions were met. Three separate logistic regression analyses were conducted using age, sex, race, insurance, lesion level, and TRAQ-SB score to predict the following dichotomous outcomes: urinary incontinence (Less than once per month vs. more frequently than once per month), skin breakdown (None vs. Any), stool incontinence (Less than once per month vs. more frequently than once per month).
Results
Sample characteristics and descriptive statistics for outcome variables are listed in Table 1. There were an equal number of male and female participants. A little over 80% of youth with spina bifida in our sample were white (82%); 16% were black; and 3% had unknown race. Only 2% identified as having Hispanic ethnicity. Our sample included almost equal numbers of children and young adults with 54% reported as under the age of 17 years and 46% being between 18 and 25 years of age. Almost half of youth and young adults with spina bifida in our sample (49%) had commercial insurance. Other types of insurance include Medicaid (39%), Medicare (5%), Tri-Care/Military (4%) and State High-Risk Plan (1%). One individual in our sample was uninsured. For the youth and young adults in our sample, thirty-five percent of youth and young adults had an Individual Education Plan (IEP).
Descriptive statistics for the demographic, clinical and outcome characteristics of our sample
Descriptive statistics for the demographic, clinical and outcome characteristics of our sample
Logistic regression results for urinary and stool incontinence and skin breakdown
Clinical characteristics of youth and young adults in our sample included lesion level and type of spina bifida. Thirty-four percent of participants had sacral lesion level, 12% low-lumber, 20% mid-lumbar, 9% high-lumbar, and 25% thoracic. The majority of participants had a diagnosis of myelomeningocele (79%), with the second most common type of spina bifida in our sample being lipomyelomeningocele (Lipoma of spinal cord), representing approximately 13%.
Listed in Table 2 are the Odds Ratio and 95% confidence intervals of the final model for urinary incontinence, stool incontinence and skin breakdown (Table 2). The model was significant in predicting urinary incontinence with the TRAQ-SB being the only significant predictor in the model. For stool incontinence and skin break down the logistic models were significantly predictive of outcomes because the Lesion Level variable was significant. The TRAQ-SB scale score was not predictive of stool incontinence or skin breakdown.
Discussion
We evaluated the relationship between self-reported self-management skills of adolescents and youngadults with spina bifida and critical clinical outcome such as stool and urine incontinence and skin breakdown to determine the predictive validity of the 11-item TRAQ-SB. In prior analyses, we demonstrated that the TRAQ-SB has very good psychometric properties, including excellent internal reliability (Cronbach
Importantly, few studies have demonstrated a relationship between transition readiness and outcomes during the transition age period of adolescence and young adulthood. This is due to the fact that self-management skills are difficult constructs to measure reliably as are patient outcomes. Because the TRAQ-SB has demonstrated good reliability, it can potentially be used to assess the complex relationship between youth self-management and disease specific outcomes. However, it also difficult to measure clinical outcomes because they are often not frequent and require both subjective measures, such as self-assessed outcomes, as well as objective measures, such as medical record documentation of skin breakdown or incontinence. To objectively document outcomes, the patient must present to the health care system and it must be recorded accurately. In patients with spina bifida, incontinence may occur commonly, but these episodes do not usually prompt the patient or family to seek clinical services, which prevents their objective documentation. Therefore, these outcomes are generally assessed using family or patient self-report. Patients and family caretakers are often reluctant to admit to their provider that they have had clinical “failures” such as stool or urine incontinence, especially when the patient’s lack of adherence contributes to the “failure.” Importantly, the patient’s response bias can also be due to their desire to please the provider, who has personally educated (or even operated on) the patient to prevent these events. Lastly, adolescents and even some young adults developmentally lack the self-reflection skills to accurately report these events. All of these factors contribute to the complexity of establishing the predictive validity of a self-management assessment questionnaire such as the TRAQ-SB and may explain the lack of relationship we found between TRAQ-SB scores and stool incontinence and skin breakdown.
More work needs to be done among youth with spina bifida to help them develop self-management skills, as is demonstrated by the few studies that demonstrate poor outcomes among young adults with spina bifida [9]. The network of 22 clinics that record data into the NSBPR is the ideal setting in which to develop new self-management measurement tools and test interventions to improve self-management. The NSBPR sites enter structured and standardized clinical, demographic and outcomes data by expert clinicians. The efforts to standardize data collection help reduce the signal-to-noise ratio so that the relationship between youth self-reported self-management skills and clinical outcomes has a better chance of emerging. We fielded the TRAQ-SB in one clinic that was participating in this network. To further refine measurement tools the effort should be expanded to multiple sites and a larger sample size. As we have documented in this study, with rigorous measurement of transition readiness and self-management and standardized measurement of outcomes, a relationship between these two constructs can be documented, however, not without limitations (e.g., we found a relationship for one outcome but not for two other outcomes).
Furthermore, the use of a brief, reliable and valid self-management tool has utility to monitor and promote the acquisition of self-management skills among youth with spina bifida. The old saw of, “What gets measured gets done,” has applicability to self-management skills. Measuring how well the youth is acquiring self-management skills should prompt discussions during clinic visits and not be lost in the many other issues that need to be discussed. We modeled the TRAQ-SB responses on the Stages of Change Model which has been used as a basis on which to assess and promote the adoption of healthy behaviors or the extinction of unhealthy behaviors and the improvement of outcomes relevant to those behaviors [15]. Using a Motivational Interviewing framework, providers can tailor their interventions to the particular stage of change of the individual youth, and work on collaboratively and set goals to incrementally adopt better skills of bowel, bladder or skin self-management. This can be done quite rapidly, even in a crowded visit for youth with complex disease. Over time the progress of the youth toward independence can be documented and new self-management behaviors can be introduced, reinforced and celebrated, creating a youth-focused and youth-friendly environment that is conducive to the adoption of increased internal motivation and self-competence, which are critical aspects of positive behavior change [16, 17].
Limitations. Our study has several limitations. The questions in the TRAQ-SB were necessarily general due to the heterogeneous nature of the self-management interventions required to maintain continence in the spina bifida population. Individuals may over or under report their self-management skills depending on how they understand the terms “necessary steps” or “recommended activities.” Research has demonstrated, however, that youth provide important and independent information on their health and health care compared to parental report [18]. Moreover, assessment of transition skills by adolescents are highly correlated with parental assessment of the same skills [19]. Secondly, as we discuss above, the measurement of the outcomes is also subject to response bias, confounding the relationship between self-management measures and outcomes. Outcomes such as urine and stool incontinence and skin breakdown may be particularly susceptible to response bias due to the stigma associated with these outcomes and because they do indicate a lack of adherence with the management plan prescribed by the treatment team, leading to under-reporting. Lastly, the time relationship between our predictor variable (self-management rating) and outcome (e.g., incontinence) is complex. We asked about self-management in the present and the outcomes were collected in the recent past (past 30 days and past year). Therefore, we are assuming current assessment of self-management skills reflects recent past self-management skills, which may not be accurate. However, our sample is primarily older adolescents and young adults, whose self-management skills are more likely to be stable.
Conclusion
Higher TRAQ-SB scores are negatively associated with bladder incontinence in youth with spina bifida. While stool continence and skin breakdown were not associated with TRAQ-SB scores, this relation is complex and may be obfuscated by either reporting bias or outcome measurement bias. To further refine the questionnaire and understand this relationship we need to field it prospectively in the SB network with larger samples. The TRAQ-SB questionnaire, however, does have value in the clinical setting to help promote the acquisition of specific self-management skills among youth with spina bifida.
Footnotes
Conflict of interest
The authors have no conflict of interest to report.
Appendix I. TRAQ-spina bifida questions