Developing and Piloting an Instrument to Prioritize the Worries of Female Youth with a Physical Disability and Mothers during the Transition to Adulthood

Abstract

Background:

The transition to adulthood is a worrying time for female youth with a physical disability and their mothers. We sought to develop an instrument to assess which transition-related worries are most (and least) worrisome to youth and mothers to inform transition preparation interventions.

Design:

A community-centered approach was used to identify and refine the most common transition-related worries of youth and mothers. A novel best–worst scaling (BWS) instrument was created in partnership with an advisory board of female youth with a physical disability and their mothers. BWS is a theory-driven survey method to identify priorities. The instrument was then pretested, refined, and piloted with youth and mothers. Findings were assessed by consultation with stakeholders.

Results:

After input from youth and mothers, 11 transition-related worries were included in the pilot instrument using an object case experimental design. Pilot testing was conducted with 22 youth and 16 mothers, including 3 mother–daughter dyads. The completion rate was 86% for youth and 100% for mothers. The instrument was sensitive enough to detect differences between youth and mothers at both the aggregate and dyad level. On aggregate, both youth and mothers worried more about health-related compared to social-related worries but differed on the prioritization of independence-related worries. Discussion of results with participants and an advocacy group confirmed the face validity of the findings.

Limitations:

Male youth and fathers were not included.

Conclusions:

The novel BWS instrument is feasible for female youth and mothers to complete independently and was valid in prioritizing worries on the aggregate and dyad levels.

Implications:

A subsequent large-scale study using the instrument may inform transition initiatives, while use among in a clinical setting may guide decision making.

Keywords

health care transition physical disability pediatric to adult care transition spina bifida cerebral palsy

Highlights

Using continuous engagement with female youth with a physical disability and mothers, we developed a best–worst scaling instrument that detects the relative importance of the worries they experience during the transition to adulthood.

The instrument was able to detect differences in relative importance of each worry and between youth and mother respondents on the aggregate level (among all participants) and on the individual dyad level (comparing individual mother–daughter pairs).

This instrument will be used on a large-scale level to inform transition preparation interventions and has potential for use in a clinic setting to inform decision making when there are 2 stakeholders (eg, youth and parents).

Both youth and mothers in the pilot study were most worried about maintaining good mental and physical health, whereas social worries such as having good friends and finding a meaningful way to spend their time were less important by comparison.

Introduction

The transition from childhood to adulthood sets the trajectory for lifelong health and well-being.¹ This life stage, recognized by professional medical societies to extend from age 12 to 26 y,² is often marked by great personal, social, medical, and legal changes. During this time, youth, supported by their parents, should increasingly assume adult roles, responsibilities, and relationships. For many, this includes preparing for and pursuing employment, becoming independent in self-care, moving out of the family home, developing mature relationships, and moving to the adult health care system.^1,3 This is considered a particularly vulnerable time for all youth due to challenges they face during this process, such as increasing delays to assuming full responsibility for their health and to independent living.^2–8

While difficult for all youth, those with physical disabilities face unique additional challenges during this transition period.^3,9,10 Many experience not only variable mobility limitations but also chronic medical conditions, executive functioning limitations, cognitive disabilities, social difficulties and barriers, and mental health conditions.^9,11–15 As such, they often face complex and compounding decisions during the transition period that are often further complicated by systemic barriers.¹⁶ Unfortunately, despite designating improving the transition to adulthood for these youth as a national priority,^3,17 youth with physical disabilities continue to experience poor outcomes during this time, such as declines in physical and mental health, social isolation, and underemployment.^18–32

Initial efforts to improve the transition of these youth focused on improving the medical transition specifically, such as establishing care with adult health care providers and becoming maximally independent in medical self-care.^33–35 Evaluation of these efforts primarily focused on process-based measures or specific components of medical transition but nonetheless overall have failed to demonstrate substantial efficacy.^36–39 However, there is an increasing recognition that health during the transition period should be considered more holistically.^3,40,41 Efforts and interventions to improve the transition process and outcomes should not exclusively focus on physical health but also consider other determinants of youth’s overall wellness within their individual, family, and social context.^3,36,41–43 They should also address person-centered priorities identified by these youth and their families themselves, such as maintaining friendships and social opportunities, minimizing chronic pain, avoiding mental health concerns, establishing romantic relationships, and providing opportunities to engage in work or recreation.^{16,41,44–52} Importantly, patients and families often experience transition and changes related to these other priorities during the medical transition, such as graduation from high school or young adult programs, insurance, guardianship, and financial implications of legal adulthood, and the increasing risk of mental health concerns during adolescence and young adulthood.^13,16,41,45 Furthermore, navigating these compounding transitions and holistic priorities involve tradeoffs. For example, strict adherence to medical programs can impede social opportunities.⁵³ A decision to file for disability limits one’s ability to get married or pursue full-time employment, which must be available, accessible, and provide adequate medical insurance.^{19,51,54–57} Such decisions are made based on the priority of the youth and family. Therefore, improving transition in a meaningful way on both a population-based and individual level requires a clearer understanding of the specific needs and priorities of families.

In this context, our primary long-term goal is to determine the relative priorities of youth with physical disabilities and their mothers during the transition to adulthood. Secondarily, our goal is to identify patterns of differences in priorities in this population, including comparing priorities of youth and mothers and identifying patterns in priorities based on factors such as youth age or disability features. As a first step toward this goal, we sought to develop and pilot test the feasibility of a novel quantitative instrument to prioritize the worries of both female youth and mothers during the transition period for use in a large-scale research study. We secondarily sought to determine the feasibility of using the instrument on an individual youth-mother dyad level as a decision support tool for future use in a clinical setting. We chose to focus on female youth as we hypothesized that the transition-related concerns of female youth and mothers would be distinct from male youth, specifically due to making decisions about sexual and reproductive health and due to their increased risk of unintended pregnancies and sexual abuse.^58–68 We focused on mothers as they typically serve as the primary caregiver and support parent for youth with physical disabilities and are more likely to discuss sexual and reproductive health concerns with their daughters compared with fathers.^69–72 A community-engaged approach was used whereby female youth and women with physical disabilities, mothers, and other advocates were involved throughout the research process. This served to ensure that the priorities of both the youth and mothers were included and the instrument was accessible to both groups.

Methods

Our goal was to develop and test a preference instrument that would identify and compare the highest priority concerns of youth and mothers during the transition to adulthood. For all phases of the study, we included youth with a physical disability affecting movement identified as female at birth ages 12 to 26 y and mothers or mother-like figures of eligible youth. Youth with physical disabilities are a highly heterogeneous population given their variable physical and cognitive abilities. To include perspectives reflecting the heterogeneity of the lived experiences of this population, we recruited youth who could answer the survey either independently or with support, mothers of youth who answered independently regardless of whether their daughter was also able to answer, and youth and mothers willing and able to answer the survey in tandem (hereafter referred to as “dyads”). The goal of including dyads was to allow for direct comparison of perspectives when disability features were the same and to test for its potential for use as a tool in a clinical setting for use on the individual family level.

Guided by the recommendations of an international task force and other relevant experts, we followed good practices for preference instrument design of using a community-engaged approach.⁷³ As an overview of the current study, we used a variety of sources to identify our research question and content areas, developed and refined our study materials, and conducted a pilot study to identify the most and least worrisome concerns about the future for female youth with physical disabilities and mothers of female youths.⁷⁴ Finally, a pilot study of the live instrument was performed.

This study was approved by the Institutional Review Board at the University of Michigan (HUM 00224330). Informed consent was obtained from all participants.

Community Engagement and Determining the Research Question

Several layers of engagement were employed from conceptualization, instrument development and refinement, pretesting and piloting, and interpretation (Figure 1). The project was initiated and questions were selected in response to pervasive worries about the transition to adulthood identified by federal agencies, advocacy groups, ongoing qualitative interviews of stakeholders, and the experience of the research team.^2,52,75,76 Namely, we sought to identify and prioritize the worries of both youth and mothers during this transition period, recognizing that worry is a strong motivating force for behavior and may provide insight into the outcomes during this time and opportunities to better support families.^77,78 Comparing concerns of youth and mothers was important for several reasons. The perspectives of these youth are underinvestigated.⁷⁹ Many acquire greater independence during this time and thus more self-determination for their behavior and ultimately outcomes.^57,80,81 However, many mothers nonetheless continue to support their daughters and may have a different perspective on the barriers to their daughter’s independence.^57,82–87 Importantly, understanding areas of divergent priorities between youth and mothers may serve to identify and address areas of conflict.

Figure 1.

Process of Instrument Development Utilizing a Community Engaged Approach.

The research team included 2 people with lived experience, 3 people with research experience, and 1 clinician who runs a clinic for youth with physical disabilities transitioning to adulthood. An Advisory Board of female youth with physical disabilities and mothers was convened and engaged throughout the process. We also worked with the Spina Bifida Association. Their leadership provided feedback on the attributes and instrument prior to the pilot study and contributed to recruitment. The Spina Bifida Association Research Advisory Council reviewed and contributed to the interpretation of pilot study results.

Phase 1: Instrument Development and Pretesting

First, a comprehensive list of all worries experienced during the transition to adulthood by female youth with a physical disability and mothers was developed. This was informed by a targeted literature review of prior transition-related studies, existing youth-focused instruments, qualitative interviews of mothers, and expert consultation with adolescent providers and the Advisory Board.^{47,56,57,87–95} This extensive list of worries or “attributes” was reviewed by the study team to determine relevancy, redundancy, and disentangle worries that were not specific to the transition period. Worries considered not specific to the transition period included current worries of youth unrelated to the future (eg, embarrassing themselves at school) and several worries of mothers about the long-term impact of supporting their daughter on other areas of their lives (eg, the impact on their romantic relationship). Thirteen attributes were selected for inclusion in the instrument.

We determined a best–worst scaling (BWS) was an ideal experimental design to prioritize and compare the worries of youth and mothers during the transition to adulthood. BWS, a stated preference method, is a rigorous, reproducible method for evaluating preferences on the same scale.^74,96 BWS is based on the economic principle of random utility theory.^97,98 Relevant to BWS, this theory postulates that when presented with multiple choices, individuals will consider tradeoffs and rationally select their perceived highest-utility choice, or a choice reflective of their perceived value or benefit, and their perceived lowest-utility choice.⁹⁹ In other words, individuals will select 2 items from a set of choices that are maximally different on an importance scale.¹⁰⁰ It requires lower cognitive burden than other approaches such as ranking, particularly important for a population that included youth and participants who may have a cognitive disability.¹⁰¹ Furthermore, unlike traditional ranking or rating methods, it provides a quantitative assessment of both priorities and their relative importance, improving the ability to discriminate between choices, and reduces scale-use bias.¹⁰⁰

The 13 attributes were incorporated into a BWS instrument using an incomplete block design. With this design, different combinations or choice sets of 5 of the 13 attributes were presented.⁹⁷ For each choice set, the participant was prompted to select the which attribute they worried about the most (the “best”) followed by which attribute they worried about the least (the “worst”) (Figure 2). Thirteen choice sets with 5 attributes each were presented, with each attribute occurring the same number of times and with other attributes the same number of times to ensure equal probability of selection.⁹⁷ Each attribute was placed in a different order within the choice set with each presentation.

Figure 2.

Example Task.

Pretesting included cognitive interviewing and codesign, assessing instrument content, presentation, comprehension, and elicitation.¹⁰² The goal of pretesting was to assess the instrument’s readability, usability, applicability, and attribute clarity and completeness for both youth and mothers.¹⁰² This, in turn, sought to improve the validity, reliability, and relevance of the iteratively improved instrument while decreasing participant burden.¹⁰² Pretesting was conducted via Zoom interviews with youth and mothers from our engagement networks and from clinics, intentionally including participants with a variety of disability experiences. Community Advisory Board members were eligible to participate. They were offered $25 for their time. During the interviews, the participants were introduced to each worry and asked to comment on the relevancy and clarity of each attribute and definition and completeness of list. Using a printed version of the instrument, they were next asked to “think aloud” as they read the introduction to the instrument and completed several tasks. Specifically, they were asked to comment on the clarity and their interpretation of the meaning of the instructions and task and explain their thoughts as they processed the tasks. The attributes and instrument were iteratively modified throughout pretesting as common themes emerged. Finally, 4 pediatric urology clinical experts reviewed the instrument and gave feedback during an in-person research meeting.

Phase 2: Instrument Pilot and Validation

A Qualtrics survey was developed based on the revised paper instrument, which included 11 attributes. The online instrument was reviewed by leadership at the Spina Bifida Association, and minor modifications were made based on their feedback. Next, participants were recruited for a pilot study via letters mailed to 50 eligible participants at our institution and an e-mail sent by the Spina Bifida Association. A flyer advertising the study was also posted in rooms of a subspecialty clinic at an academic medical center. Both the letter and flyer included a QR code that linked to the survey so participants could complete the instrument on their own devices at a convenient time and place. Participants were eligible to enter a lottery for one of two $50 prizes. Eligible female youth with a physical disability and eligible mothers were invited to take the instrument together one after the other if feasible (eg, as a dyad) but were also allowed to participate independently. At the end of their survey, youth were asked if they completed the survey independently or with varying degrees of assistance.

Best–worst (B-W) scores were calculated using aggregated scoring. The dependent variable in BWS is the participant’s perception of the most and least worry in each choice set.¹⁰³ B-W scores were calculated both at the aggregate and individual level for each worry attribute. We subtracted the number of times respondents selected an attribute as “least worried” from the number of times selected as “most worried” and then divided by the total number of times the attribute appeared in the survey. The resulting B-W score ranges from +1 to −1, with higher scores signifying greater prioritization of the attribute.

B-W scores are highly correlated with conditional logistic regression estimates at characterizing the relative importance of attributes.⁹⁷ A conditional logistic regression was performed in aggregate to confirm the priority scores calculated with this method corresponded to the calculated B-W scores to confirm the validity of the results and appropriateness of this statistical method in the future large-scale study. We assumed a sequential best–worst decision-making process, meaning each survey item included 2 choice sets. First, the best attribute was selected from the entire choice set (5 attributes), and the other attributes were not selected (each attribute assigned a value of 1 or 0). Next, the worst attribute was selected from the remaining choices (4 attributes), and the other attributes were not selected (each attribute assigned a value of −1 or 0). In the model, the best and worst data were pooled, and the choice (selected or not selected) was the dependent variable. Effects coding was performed to compare the effect of each attribute. The coefficients for each attribute were interpreted as a priority score. Pearson’s rho assessed B-W score profile concordance between mothers and daughters within each dyad.¹⁰⁴ We conducted pseudo couple analysis to assess the significance of dyadic concordance.¹⁰⁵ Profile correlations testing for similarities on the individual dyad level were computed with adequate reliability as each individual’s profile contains 11 worry attributes, exceeding the recommended minimum of 8 items.¹⁰⁶ Analyses were performed using Stata BE version 18 (StataCorp, College Station, TX, USA).

Results

Pretesting Results

From May to September 2023, 5 mothers (ages 41–49 y) and 4 youth (ages 15–21 y) participated in pretesting. All expressed enthusiasm for the instrument development. They all felt the transition to adulthood is a source of anxiety for both youth and mothers and that their concerns need to be better understood to improve care and relevant interventions.

Nine iterations of the list of attributes, attribute definitions, and supporting pictures were tested to address clarity, comprehension, relevance, and completeness for both adolescents and mothers (Table 1). One attribute was added based on feedback (“avoiding chronic pain”) while others were consolidated as pretesting participants felt they were overlapping (eg, “job and hobbies,” “school and job absences,” and “accepting mentors” were consolidated to “having a meaningful way to spend your time”; “fun friendships” and “close friendships” were consolidated to “having good friends”). Participants preferred attributes to be stated in the positive, especially if stigmatizing (eg, “having good mental health” not “avoiding mental health problems”). Youth participants desired inclusion of representative pictures to be included in the attribute definitions. As pictures were added and refined, youth expressed that icons were more relatable and inclusive than pictures of youth with physical disabilities.

Table 1.

Attribute Refinement.

Attribute	Representative Quote	Revised Attribute	Description
Physical independence	“I didn’t realize how disabled I was until I moved away from home.” (Youth 40)	Taking care of yourself	Being able to do things for yourself like getting dressed, cooking, transportation, or living on your own
Healthy romance	“Everyone no matter their ability deserves to have that kind of intimacy if they want it.” (Mom 39)	Finding a life partner	Finding a life partner or companion such as a boyfriend, girlfriend, husband, or wife
Fun friendships	“The closest friend I have I hang out with her a lot and I sometimes tell her a little bit about what I have going on. . . .” (Youth 4)	Having good friends	Having good friends you can trust and enjoy spending time with
Close friendships	“I want more close friends . . . you can trust them, tell them your secrets and they won’t blob it out.” (Youth 16)	—
Personal safety	“Her sense of judgment is off, not knowing who is genuine, I worry about her being taken advantage of.” (Mom 17)	Staying safe from abuse	Staying safe from people who may try to hurt you
Money problems	“I feel like a lot of people, including myself, aren’t fully able of understanding all of that cognitively.” (Youth 39)	Dealing with the transition to legal adulthood	How to figure out insurance, guardianship, and finances that change when you become an adult
Care needs	“Complete independence is not a guarantee for children that become adults with physical disabilities . . . .” (Mom 16)	Finding someone to support you if your parents can’t	Finding someone to help you with any needs when your parents can’t help anymore
Health care	“I experienced the drop-off between pediatric and adult care . . . it was a big problem.” (Youth 40)	Moving into the adult health system	Finding good doctors and nurses in the adult hospital to care for you
Mental health	“People who are disabled who in my opinion are more likely to be depressed.” (Youth 40)	Having good mental health	Avoiding depression or anxiety
Physical health	“If you are in pain, then you can’t do all those other things . . . .” (Mom 2)	Staying physically healthy	Staying well and out of the hospital
Job and hobbies	“This can cover a lot, whether it be going to church or hobbies or whatever you do over time.” (Mom 16)	Finding a meaningful way to spend your time	Finding a supportive job, volunteer work, hobby, or other ways to spend your daily adult life
School and job absences	“This is related to physical health.” (Mom 39)	—
Accepting mentors	“It’s important, but [other people] might not know it’s important.” (Youth 39)	—
		Avoiding chronic pain	Not hurting or having pain most days

Initially, the question was framed in the third person to minimize negative emotional responses and allow the attributes to be identical for both youth and parents (eg, both in the third person). Participants were asked to consider “Jane” or “Jane’s mom” who were “a lot like them” and select what they thought “Jane” or “Jane’s mom” worried about the most and the least. However, despite several iterations of clarifying the instructions, this concept remained confusing for both participant groups. Ultimately, the question stem was changed to the first person, and mirrored attributes were developed (eg, “taking care of yourself” for youth and “taking care of herself” for mothers) (Figure 2).

Once saturation was met, meaning no new significant changes were made by participants, the instrument was shared with both a clinician group who care for these youth and the Spina Bifida Association’s leadership. Both stakeholder groups felt the study was highly relevant. They suggested several minor syntax modifications for clarity, resulting in the final pilot instrument (Appendix A).

Pilot Results

A total of 38 participants, including 22 youth and 16 mothers, completed the pilot survey. They included people with a variety of disability types, mobility aids used, and races (Table 2). We noted post hoc that 2 youth completed the instrument, including the prescreening questions stating they met the age criteria, despite being older than the age cutoff for participation. Their responses were included in the analysis as the goal was to test the analytic approach and not report generalizable results. Of the youth participants, 19 completed all portions of the BWS questions, with 3 missing data (eg, choosing a “best” but not a “worst” option). Of the mother participants, all 16 completed all portions of the BWS questions, with no missing data. The instrument was sensitive enough to identify significant differences in best–worst scores on aggregate (eg, youth and mothers together) as well as differences between scores of adolescents compared with mothers (Figure 3). As anticipated, the calculated B-W scores corresponded to the priority scores estimated by the logistic regression (Appendix B). Overall, there was agreement between youth and mothers over the relative importance of health-related concerns, namely, “having good mental health,” “staying physically healthy,” and “avoiding chronic pain.” By comparison, social-related concerns such as “having good friends” and “finding a meaningful way to spend your time” were less important. Disagreement was noted over the relative importance of independence- and safety-related concerns, including “taking care of yourself,” “finding someone to support you if your parents can’t,” and “staying safe from abuse,” which mothers considered more important, as well as over “finding a life partner,” which youth considered more important.

Table 2.

Pilot Study Participant Demographics.

Participant	Youth Participants	Daughters of Mother Participants
Youth age, y	21.4 (range 14–32)	17 (range 13–25)
Disability type of youth
Spina bifida	3	2
Cerebral palsy	5	4
Spinal cord injury	5	1
Other	5	8
Unknown	4	1
Race of youth
White	10	11
Black or African American	8	4
Asian	1	0
Hispanic or Latino	1	0
Unknown	2	1
Mobility aid(s) used by youth
Wheelchair	7	4
Walker	1	1
Crutches or cane	1	2
Ankle-foot orthosis or similar	3	0
Shoe lift	3	1
None	5	9
Unknown	3	1

Figure 3.

Youth vs. Mother Aggregate Best-Worse Scores with Standard Errors.

Three dyads, or daughter–mother pairs, completed the survey in the same setting. One daughter filled it out completely independently, one “mostly” completed it independently with “some help,” and the last had someone help her fill it out. The remainder of youth who participated in the study completed it independently. Within-dyad comparison was feasible. Overall, preferences of daughter–mother dyads were more similar than different, especially when compared with randomly selected youth–mother pairs of those who participated independently (r = 0.595 vs r = 0.041, respectively). Nonetheless, differences were noted between youth and mothers in each dyad, suggesting that the surveys reflected the preferences of each party and that the mother did not complete the instrument on behalf of her daughter.

Results were shared with 3 participants, including 1 dyad and 1 youth, and the Spina Bifida Association Research Advisory Council, which included multidisciplinary providers, researchers, and people with lived experience. All members felt that the results overall reflected what they perceived as community priorities and sources of conflict in families. They noted that mental health conditions are highly prevalent in youth and young adults with disabilities and were glad that there seems to be awareness of this in the community.^23,24,27 They also affirmed that the independence-related worries are often a source of conflict between parents and youth and as such found their discrepancies in priorities in this reflective of real differences.^87,107 The participants and the Research Advisory Council were surprised that “having good friends” was a low priority for youth and mothers given published data suggesting these youth often struggle with social isolation.^18,28,55 The youth participants expressed that they felt this was a big concern for “other people,” but not for them. After discussions with these stakeholders, the consensus was that the attribute should be changed to “having close friends,” as they perceived having friends to confide in becomes more important with age. All community members felt the study was important as it quantified the relative priorities and degree of relative priorities of the community and could help support or promote initiatives addressing these worries. The community endorsed expansion of this study to a national study.

One adolescent shared that the instrument was difficult for her to fill out due to her visuospatial limitations. Namely, she struggled to spatially visualize both the “most worried” and “least worried” column. She thought she likely forgot to select a “least worried” choice at times due to this and was not forced to answer as Qualtrics only forces 1 answer. After probing her suggestions to improve the accessibility of the instrument, the study team sought to add lines between each attribute, use colors to highlight both columns, and force respondents to answer on both columns to proceed to the next question. As these were not features available with Qualtrics, a software engineer and PhD student developed code to add these functionalities. Iterative revisions were reviewed with the research team, which included a person with low vision, and a team of pediatric research experts until a final version was approved.

Discussion

Successfully achieving greater adult roles, responsibilities, and relationships while maintaining health and well-being as a youth is thought to set a positive trajectory for adulthood.^1,32 Despite national attention to improving this transition process, outcomes for female youth remain poor.^{21,22,57,108–111} A holistic understanding of the worries of these youth and their mothers during this time is necessary to inform family-centered interventions that could improve their outcomes. In this study, we developed and piloted an instrument to prioritize and compare the worries of these youth and their mothers during the transition to adulthood. Using a community engaged approach, we identified 11 primary worries of youth and mothers that often involve tradeoffs (eg, finding a life partner vs staying safe from abuse). The pilot study demonstrated that the instrument was sensitive enough to detect similarities and differences in the priorities of youth and mothers on the aggregate level as well as on the individual dyad level. In addition, we identified key factors for improving instrument accessibility for youth with physical disabilities. This will next be used on a large scale to better understand the similar and discrepant priorities of youth and mothers as well as how priorities may differ within this population, such as by the youth’s age or disability features. Ultimately, this can serve to inform transition-related interventions. This and other dyad instruments may also have value in a clinical context for comparing the priorities of youth and parents for clinical counseling and decision making.

The pilot results informed hypotheses for the upcoming larger national study. There was overall consensus between youth and mothers on the prioritization of health-related worries and the relative deprioritization of social-related worries. This suggests that, for this pilot cohort, if health-related therapies or regimens conflict with social opportunities, they would prioritize their health (eg, attending therapy instead of extracurricular activities). Perhaps this reflects the increasing awareness of the mental and physical health decline many youth with physical disabilities experience.^{20–27,29,32} While evidence shows that social isolation as well as underemployment for these youth is common, for this pilot cohort, it was nonetheless a lower priority by comparison.^{18,28,54,55,58} We hypothesize that health-related worries will continue to be prioritized over social-related worries by youth and mothers in the national study. There was discrepancy between youth and mothers over independence-related worries. Similarly, other studies have also noted that while parents may consider gaining independence a priority, youth may not.^{15,87,107,112} However, there is some evidence that it may become an increasing priority for youth with age.⁸¹ We anticipate that the large-scale study will demonstrate a similar discrepancy for school-aged youth, but the discrepancy will be significantly less for youth who have graduated as we anticipate they will be actively working toward independence. Indeed, we anticipate finding significant differences in priorities overall based on whether the youth has graduated, with those who are younger or have not completed their education demonstrating different priorities than those who are older or have graduated and are further in the transition process. Despite qualitative findings suggesting mothers strongly hope their daughters will find a life partner, this was the lowest priority for mothers, whereas it was a higher priority for youth. Conversely, mothers worried more about safety than youth did. Similarly, other studies have reported the tension between youth who are interested in having a romantic relationship and parents who are concerned about their safety.^2,30 We hypothesize that these discrepancies will continue to be observed in the large-scale study. Finally, we hypothesize that we will continue to identify differences among dyads, but they will show greater correlation than random adolescent–mother pairs and aggregate analysis.

Continuous stakeholder engagement was critical for developing a relevant, complete, and accessible instrument for these youth with physical disabilities and their parents. These youth are a heterogenous group with variable physical disabilities, some of whom also experience variable cognitive disabilities, executive functioning limitations, and visuospatial processing difficulties. Nonetheless, the key worries as identified by youth and parents of youth with variable disability features during pretesting and confirmed during the pilot overlapped. The large-scale study will be able to discern if there are any differences in the relative priority of these worries based on the youth’s disability features. Many iterations of the attributes and instrument itself were made to ensure the final instrument was maximally accessible. Key modifications included keeping language throughout the instrument as simple as possible and adding explanatory pictures to the attribute descriptions. In addition, with the assistance of a researcher with a background in software engineering, we were able to enhance the appearance of the tasks and force both a “most” and “least” answer to improve functionality for those with visuospatial processing and executive functioning limitations. While labor intensive, the research team and all stakeholders agreed it was critical to include the youth voice for a population whose voice has been historically marginalized and may be reluctant to voice an opinion contrary to that of their parents.^79,113,114

The strengths of this study include the inclusion of youth of various ages, disabilities, and abilities; mothers; and other community members in the development, refinement, and testing of this instrument and the perceived importance of the question. In addition, we were able to detect differences on the aggregate and individual dyad level. However, the study is not without weaknesses. First, by design, only female youth and mothers were included. Male youth and fathers may have different worries and priorities. Although pilot participants included youth and mothers of youth with a good representation of different disability types, feedback was obtained from an advocacy group for only 1 disability type. It is possible that accessibility concerns specific to other disabilities were missed. We will offer the option to take the survey by phone or Zoom with a member of the study team to help with this. Initial attribute identification was inclusive of a targeted literature review, a recent qualitative study, and study team input that was inclusive of the perspective of adolescents with a variety of physical disabilities and their families. Therefore, we do not anticipate that any significant attribute is missing, although this is a possibility. We will continue to offer the option to write in any worries that are not included on the list of attributes to monitor deficiencies. In addition, we assume that each participant’s answers reflected their own priorities but cannot verify as participants took the survey online on their own time. For dyad respondents in particular, it is possible that their responses could have been influenced by one another. One daughter reported completing the survey completely independently, while another completed it mostly independently with some help, and the last required help to complete it. Although the priorities of each dyad were more alike than random mother–daughter pairs, the dyad youth and mother responses also reflected distinct differences, suggesting the youth perspective was included at least in part. The future large-scale study will determine if this finding remains consistent among a larger number of dyads and if mothers are unduly influencing the survey response of the youth or if similarities may reflect true familial priorities (eg, mothers and their daughters sharing certain priorities). Finally, the strength of this methodology is that it rigorously identifies relative priorities, but not absolute priorities. As such, this facilitates discerning which worries are the highest priority to participants but not how often or to what degree they worry.

There was strong support of all stakeholders to expand this to a national study. Stakeholders felt this will help advocate for programs, funding, and other initiatives that will help improve outcomes during this transition period. In addition, stakeholders commented that understanding the priorities of youth and mothers may give insight to where they are likely to make tradeoffs (eg, if social considerations are prioritized over physical health in the larger study), which providers could consider when making care decisions. Understanding where discrepancies between youth and mothers are common, such as over independence-related worries in these preliminary findings, can also identify key targets to address in family-centered transition interventions.

This study accomplished the primary objective to develop and test the feasibility of a preference instrument to prioritize and compare the worries of youth with physical disabilities and mothers during the transition to adulthood. The secondary objective of establishing the feasibility of analyzing the instrument on an individual dyad level was also achieved. There are several possible future implications of this, for both this specific dyad instrument and similar instruments. First, it can be used to inform real-life priorities in the setting of interventions. This instrument could augment existing transition preparation interventions by determining where priorities of female youth and parents during this transition are similar and where they are discrepant. This information could be used to facilitate conversations, provide educational materials, and better support youth and parents. Second, a dyad instrument could be used in making clinical decisions. Although youth should be increasingly involved in decisions regarding their care, it can be practically difficult to execute. A dyad stated preference instrument could facilitate shared decision making that ensures elicitation and consideration of both the youth and parent priorities. Finally, this approach could be used in the research setting such as this to compare between other relevant stakeholder groups (eg, couples, patients, and providers).

Conclusion

This study demonstrates the feasibility of administering a stated preference survey to adolescent girls with a physical disability and mothers to prioritize their worries during the transition to adulthood. Stakeholder feedback was critical to refine the instrument to maximize clarity and functionality. Preliminary findings suggest that the large-scale study results will serve to identify opportunities to better support the priorities of youth and mothers during this time both by ensuring interventions align with their priorities and by demonstrating where attention is needed to address areas of discrepancy. In addition, a novel application of BWS to identify correlation and differences in priorities among dyads has implications for use of this approach in clinical care to inform decision making.

Supplemental Material

sj-docx-1-mpp-10.1177_23814683261457144 – Supplemental material for Developing and Piloting an Instrument to Prioritize the Worries of Female Youth with a Physical Disability and Mothers during the Transition to Adulthood

Supplemental material, sj-docx-1-mpp-10.1177_23814683261457144 for Developing and Piloting an Instrument to Prioritize the Worries of Female Youth with a Physical Disability and Mothers during the Transition to Adulthood by Courtney S. Streur, Dalia Elased, Jodi M. Kreschmer, Rebecca Parten, Hamoud Alhazmi, Brittany Gay, Ashley Duby, Henrike L. Schmalfuss, Jenna Goldstein, Grayson N. Holmbeck, Lisa A. Prosser and John F. P. Bridges in MDM Policy & Practice

Supplemental Material

sj-pptx-1-mpp-10.1177_23814683261457144 – Supplemental material for Developing and Piloting an Instrument to Prioritize the Worries of Female Youth with a Physical Disability and Mothers during the Transition to Adulthood

Supplemental material, sj-pptx-1-mpp-10.1177_23814683261457144 for Developing and Piloting an Instrument to Prioritize the Worries of Female Youth with a Physical Disability and Mothers during the Transition to Adulthood by Courtney S. Streur, Dalia Elased, Jodi M. Kreschmer, Rebecca Parten, Hamoud Alhazmi, Brittany Gay, Ashley Duby, Henrike L. Schmalfuss, Jenna Goldstein, Grayson N. Holmbeck, Lisa A. Prosser and John F. P. Bridges in MDM Policy & Practice

Supplemental Material

sj-pptx-2-mpp-10.1177_23814683261457144 – Supplemental material for Developing and Piloting an Instrument to Prioritize the Worries of Female Youth with a Physical Disability and Mothers during the Transition to Adulthood

Supplemental material, sj-pptx-2-mpp-10.1177_23814683261457144 for Developing and Piloting an Instrument to Prioritize the Worries of Female Youth with a Physical Disability and Mothers during the Transition to Adulthood by Courtney S. Streur, Dalia Elased, Jodi M. Kreschmer, Rebecca Parten, Hamoud Alhazmi, Brittany Gay, Ashley Duby, Henrike L. Schmalfuss, Jenna Goldstein, Grayson N. Holmbeck, Lisa A. Prosser and John F. P. Bridges in MDM Policy & Practice

Footnotes

Acknowledgements

The authors would like to acknowledge the Spina Bifida Association leadership, Judy Thibadeau, RN, MN, Laura Carlson, PhD, and Juanita Panlener, for their contributions to this work and support with recruitment. The authors would also like to thank the members of the Spina Bifida Association Research Advisory Counsel for their input on the study results.

ORCID iDs

Courtney S. Streur

Henrike L. Schmalfuss

Grayson N. Holmbeck

Funding

The authors disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: Financial support for this study was provided by the Eunice Kennedy Shriver National Institute of Child Health and Human Development under Grant 1K23HD10598701A1 (Streur) and by the Burroughs Wellcome Fund under Grant Innovation in Regulatory Science Award (Bridges). The funding agreement ensured the author’s independence in designing the study, interpreting the data, writing, and publishing the report.

Declaration of Conflicting Interests

The authors declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Ethical Considerations

Institutional Review Board approval from the University of Michigan was obtained prior to initiating this study (HUM 00224330).

Consent to Participate

Oral consent was obtained from participants who participated in interviews with the research team. Implied consent was assumed for survey participants. The first page of the survey was a consent form. At the bottom of the consent, participants had the option to “agree” and continue with the survey or decline, in which case the survey ended.

Consent for Publication

We do not use identifiable information in this study.

Data Availability Statement

The iterations of the survey items and deidentified survey results are available upon request.

Supplemental Material

Supplementary material for this article is available online.

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