Inclusive measure development: amplifying the voices of adolescents and young adults with spina bifida in a new measure of benefit-finding and growth

Abstract

Objective

Benefit-finding and growth is an important process across a range of medical populations. However, it has been understudied in the context of lifelong chronic conditions, such as spina bifida (SB). This study aimed to develop a new measure of benefit-finding and growth for youth with SB, confirm its factor structure, and examine its psychometric properties.

Method

To generate items for the new measure, 20 adolescents and young adults with SB completed qualitative interviews regarding their experience of living with SB. Interviews were coded for benefits. Questionnaire items were generated from these benefits, and an expert panel refined the wording of these items. The resultant 31-item measure was shared with six of the 20 participants for feedback and then piloted among 251 youth with SB. The factor structure of the measure was confirmed and reliability and convergent validity were assessed.

Results

Both a one- and four-factor structure were supported. The four factors include: Life Perspectives and Priorities, Personal Characteristics and Traits, Connections and Opportunities, and Problem Solving. Higher total and factor scores represent greater benefit-finding and growth. The measure demonstrated excellent internal consistency (α = 0.95). The new measure also showed significant positive correlations with optimism, positive affect, and life satisfaction.

Conclusions

This study produced a measure of benefit-finding and growth for youth with SB. Clinically, information about what youth with SB perceive to be their areas of strength and growth from their condition provides crucial insight into which factors to enhance in this population.

Spina bifida (SB) is a neural tube defect that occurs when the embryonic neural tube fails to close early in prenatal development. This opening exposes the neural tube to the amniotic fluid, which is ultimately toxic to the spinal cord and causes neurodegeneration in utero (Copp et al., 2015). The clinical presentation and degree of impairment vary widely and depend on the type of SB and the location of the opening (i.e., lesion) on the spinal column (CDC, 2021). The most common and severe form of SB is myelomeningocele in which the lesion is open and the spinal cord and nerves develop externally in a fluid-filled sac on the back area (Copp et al., 2015). Alternatively, in meningocele SB, the lesion is closed and the lesion contains meninges and cerebral spinal fluid only (CDC, 2021). In terms of the lesion level, the most common location is the lumbar spine, followed by sacral, thoracic, and cervical (CDC, 2021). Condition-related deficits occur below the level of the lesion. Therefore, the higher the level of the lesion, the more significant the impairment, as well as the greater the risk for complications (Gober et al., 2022).

Depending on the type of SB and lesion level, SB can be associated with a broad range of complications, including bowel and bladder incontinence, pressure injuries, and hydrocephalus requiring shunting and shunt revisions. Orthopedic abnormalities such as limb weakness can impair or entirely disrupt ambulation. With its impact on neurocognitive functioning, SB is also associated with difficulties with attention, executive functioning, social skills, and independent functioning (Stiles-Shields et al., 2019). Psychosocially, youth with SB are at higher risk for depression, social problems, and lower health-related quality of life than their typically developing peers (Copp et al., 2015; Kritikos et al., 2020).

Despite the stressors and challenges that individuals with SB face, they exhibit considerable resilience. For example, in emerging adults with SB, resilience is seen in the areas of identity development, quality of social supports, and less alcohol and substance use (Holmbeck & Kritikos, 2022). Yet, most psychosocial research in youth with SB has focused exclusively on negative outcomes. Pediatric psychologists should aim to accurately represent and address the full landscape of psychosocial functioning in those with SB, including assessment of strengths. Clinically, understanding positive adjustment in adolescents and young adults with SB could inform the development of successful prevention and intervention efforts for youth with SB. Specifically, identifying the coping skills that resilient youth use naturally could elucidate factors to target or enhance in youth who are struggling.

Benefit-finding and growth involves identifying benefit or positive change that has occurred because of a stressor, illness, or traumatic event (Park, 2009). The cognitive process of benefit-finding and growth is rooted in theories of coping with stress (Janoff-Bulman, 1989) and observations that highly distressing events involving suffering can also lead to positive changes and growth in an individual (Calhoun & Tedeschi, 2014). Such changes may include changes in the self, relating to others, and life philosophy (Calhoun & Tedeschi, 2014), among others (Kritikos et al., 2021b). Benefit-finding and growth shows promise as a process related to positive psychological outcomes in other pediatric health populations (Kritikos et al., 2021b). Evidence from cross-sectional, longitudinal, and meta-analytic studies also supports associations between benefit-finding and growth and psychosocial outcomes such as optimism, positive affect, life satisfaction, and a host of other positive outcomes (Kritikos et al., 2021b). Based on a systematic review of the current literature on benefit-finding and growth in pediatric medical populations, researchers recommend that future research on benefit-finding and growth begin with qualitative interviews to determine perceived benefits in the words of individuals from a new population, and establish the factor structure of a measure prior to use in a novel population (Kritikos et al., 2021b).

Benefit-finding and growth in the context of SB requires multiple considerations as a novel area of study. First, SB is a lifelong condition. Many individuals with SB are diagnosed in utero and do not know a life without SB (Copp et al., 2015). The lifelong, chronic nature of SB differs from the more typically discrete “events” or singular medical traumas, where benefit-finding and growth has been studied as a change from before and after an event. Nonetheless, the centrality, pervasiveness, and chronicity of SB likely only increase the importance of benefit-finding and growth for two reasons. First, research shows that the more central to one’s identity that a stressor is, the more benefits that can be drawn from the event (Watanabe, 2019). Second, chronic disease might provide individuals with repeated opportunities to remind themselves of the positive changes or growth that is possible in the context of their condition (Park, 2009).

To our knowledge, there is one prior study on benefit-finding and growth in the context of SB (Kritikos et al., 2021a). A survey study with young adults with SB (Kritikos et al., 2021a) adapted an existing self-report measure of benefit-finding that was developed for women with breast cancer (Tomich & Helgeson, 2004) for use with individuals with SB. Findings supported the experience of benefit-finding and growth among a nationally representative sample of adults with SB, with respondents reporting benefits related to their personal strengths, interpersonal relationships, and life philosophy due to SB (Kritikos et al., 2021a). However, psychometric properties of the measure, including the factor structure were inconsistent. Thus, Tomich & Helgeson’s (2004) measure developed for women with breast cancer may have limited utility with adults with SB. Instead, an approach that focuses on the distinct experience of adolescents and young adults with SB, centers their voices, and accounts for the distinctive features of SB is likely to yield a more appropriate, comprehensive measure of benefit-finding and growth.

This two-part study aimed to develop a self-report measure of benefit-finding and growth for adolescents and young adults with SB, confirm the new measure’s factor structure, and validate the measure. In our measure development process, we follow Holmbeck & Devine’s (2009) measure development and validation guidelines, as well as recommendations from a systematic review of benefit-finding and growth in pediatric health populations. Specifically, we partnered with our target population in the creation of the measure, used qualitative methods, and began with a psychometric study (Kritikos et al., 2021b). In part one of the study, we conducted individual qualitative interviews with adolescents and young adults with SB to gain insights into their positive lived experience of SB. Based on these data, possible items were generated for the measure. Given the qualitative and exploratory nature of this portion of the study, the goal was to identify and quantify the benefits that adolescents and young adults with SB would describe.

In part two of the study, we piloted the measure in a national sample of adolescents and young adults with SB and confirmed the factor structure. Based on the benefits identified in part one’s qualitative interviews and the themes around which they clustered, we hypothesized that in addition to a single factor, a four-factor structure would fit the data, with the four factors including life perspectives and priorities, personal characteristics and traits, connections and opportunities, and problem-solving.

In addition, we examined the reliability (internal consistency) and validity (concurrent validity with measures related to the construct of benefit-finding and growth) of the new measure. Specifically, we examined the concurrent validity of the new measure to three constructs that have shown positive correlations to benefit-finding and growth in the literature (Kritikos et al., 2021b): optimism, positive affect, and life satisfaction. We hypothesized that we would find evidence of positive associations between our new measure of benefit-finding and growth and each of these three constructs.

Part one methods

Participants

Participants in part one of the study, which aimed to generate possible items for the new measure, were recruited from the Illinois Spina Bifida Association’s (ISBA) social media and email accounts. Interested participants reached out to study investigators via email and were screened for eligibility. Eligible participants: (a) had SB, (b) were between the ages of 15 and 30 years, and (c) were able to meet via zoom for a qualitative interview and answer survey questions without the assistance of others (ensuring privacy and honesty in responses). Compensation was provided via a $20 Amazon electronic gift card.

Procedure

Part one participants were recruited between February and May 2021. Consistent with University Institutional Review Board (IRB) compliance and previous national online surveys conducted with individuals who have SB (Kritikos et al., 2021a), participants were consented via digital waiver of documented consent. For participants ages 15–17 years old, parents provided digital waiver of consent, and adolescent participants provided assent.

In part one of the study, semistructured, open-ended qualitative interviews were conducted by the lead author with 20 adolescents and young adults with SB to explore benefit-finding and growth. The interview began broadly with a narrative approach, wherein participants were prompted to reflect on their comprehensive experience of SB rather than being forced to focus solely on positive aspects (Tedeschi et al., 2018). Participants were then asked specifically about the challenges of living with SB (e.g., “Tell me some of the toughest/hardest/most challenging parts of living with spina bifida?”) and any benefits gleaned from living with SB (e.g., “Are there any ways that spina bifida has positively affected your life?”). This approach gave the respondents a collaborative role in determining the direction of the interview and allowed the interviewer to capture the ideographic experiences of adolescents and young adults with SB (Cohen et al., 1998). Interviews lasted approximately 20–30 minutes. All interviews took place via zoom and were audio recorded and transcribed verbatim by a third-party transcription service. Demographic and medical information, including age, gender, race and ethnicity, education, employment status, type of SB, shunt status, and lesion level, were collected via self-report questionnaire in order to characterize the sample.

Data analytic plan

All qualitative interviews were double coded by a coding team, including the lead author and two doctoral-level graduate students (authors A.M.W. and O.E.C.). Qualitative analyses followed the overarching principles outlined by Wu et al. (2016) with reflexive thematic analysis as our guiding framework (Braun & Clarke, 2019). Specifically, the coders read the entire interview and focused on themes that emerged from responses to the interview questions: “Tell me some of the ways in which SB has positively impacted your life,” “How do you tend to think of SB generally” and “Are there any positives or any ways that SB has positively affected your life?” Every response affirming a benefit was recorded and added to a new emergent category. The team of coders met after coding interviews in batches of approximately five interviews at a time to review emergent benefits, discuss wording of the benefit, and come to a consensus on the collapsing of various responses into one benefit category (e.g., problem-solving ability). In this way, the “code book” was built in an iterative process as new “codes” (i.e., benefits) emerged. These codes then informed selection of items for the self-report measure. Consistent with Holmbeck & Devine’s (2009) measure development guidelines, item selection for the new measure was based on: clinical experience, relevant theories (e.g., Park, 2009), empirical literature (e.g., Kritikos et al., 2021b), rational deduction, related instruments (e.g., the Benefit-Finding Scale; Tomich & Helgeson, 2004), consultation with experts, and interviews with the target population.

Following initial item generation and data saturation after 14 interviews, a panel of four experts, consisting of prominent researchers in the field of SB and benefit-finding and growth (authors C.S.-S., V.H., G.H., as well as an additional collaborator), met with the lead author (T.K.K.) to review each of the proposed items and provide feedback on phrasing, consolidation of items, literacy and reading level issues for the target population, and appropriate instructions (Holmbeck & Devine, 2009). The resultant 31-item measure was then shared with the six remaining participants in part one of the study in the following manner: first, these six participants completed the same interview as the earlier 14 participants, providing responses to questions about their experience of SB. Then, the interviewer (T.K.K.) shared the draft of the 31-item measure and elicited feedback from the interviewees regarding the content and phrasing of the items (per guidelines from Holmbeck & Devine, 2009).

Considering positionality and reflexivity, the lead author was a postdoctoral fellow specializing in SB-related research. At the time of the study, she had completed her internship in pediatric psychology and worked with a variety of chronic health populations. However, she was not involved in any clinical work with SB patients and has no personal experience with SB. To promote rigor in our qualitative research, the following strategies were used (Nowell et al., 2017; O’Brien et al., 2014). Collaborative coding meetings among the three coders included debriefing of interviews, discussion of potential biases, discrepancies, and self-evaluation to enhance reflexivity. Codes were added to the codebook until saturation was reached. During the expert panel meeting, each potential measure item was presented alongside representative quotes of participants’ perceived benefits from the participants across the dataset. This panel of experts also brought additional varied perspectives from their experiences working with SB populations that were acknowledged and discussed in the expert meeting.

Part one results

Sample characteristics

Part one included 20 adolescents and young adults with SB. The sample had a mean age of 25.55 years (SD = 3.75; Mdn = 26.0; range: 15–30 years) and predominantly identified as female and White, non-Hispanic/Latine, and had myelomeningocele type of SB. Table 1 provides descriptive information about this sample. Additionally, of the subsample of six part one participants who provided feedback on the measure, the mean age was 26.17 years (SD = 3.65; range: 20–30 years). Five of the six participants were female. Two of the participants identified as Asian; one participant identified as non-White Hispanic, one participant identified as White and Hispanic, and two participants identified as White, non-Hispanic.

Table 1.

Demographic and medical characteristics of 20 interview participants.

Characteristic	N (%)
Age, M (SD; range)	25.55 (3.75; 15–30)
Gender
Male	6 (30%)
Female	14 (70%)
Race and ethnicity
African American	0 (0%)
Asian	2 (10%)
White	12 (60%)
Latine	6 (30%)
Highest level of education
Some high school	3 (15%)
Finished high school	4 (20%)
Business or technical school	2 (10%)
Some college	6 (30%)
Finished college	3 (15%)
Attended graduate or professional school	1 (5%)
Received a professional degree	1 (5%)
Employment status
Unemployed	9 (45%)
Volunteer	1 (5%)
Student	1 (5%)
Disability from work	1 (5%)
Employed, part time	4 (20%)
Employed, full time	4 (20%)
SB type
Myelomeningocele	15 (75%)
Meningocele	1 (5%)
Lipomeningocele	1 (5%)
Lipmyelomeningocele	1 (5%)
Lipoma	1 (5%)
Unsure	2 (10%)
Shunt status
Yes	15 (75%)
No	5 (25%)
Shut revision
Yes	14 (70%)
No	6 (30%)
Number of revisions, M (SD; range)	2.00 (1.96; 0–6)
Lesion level
Lumbar	13 (65%)
Thoracic	1 (5%)
Unsure	5 (25%)
Missing	1 (5%)

Interview responses

Interviewees endorsed a range of possible benefits to living with SB. Based on the responses from the first 14 interviews, coders identified 22 possible benefits. Some of these benefits were iterated in as many as 12 out of 14 interviews (i.e., problem-solving abilities) while others were only mentioned by one respondent (e.g., courage, spirituality). The expert panel met and expanded these 22 items into 31 items, primarily by breaking up items that included compound statements (e.g., the item “…has helped me to live in the moment and appreciate good moments” was divided into two items, one capturing living in the moment and one capturing appreciating good moments). Additional changes were made to simplify language and vocabulary (e.g., instead of “…has helped me to enjoy helping or advocating for others,” item was rephrased as “helping others”). Priority was given to comprehensiveness of the measure; therefore, even benefits that were only mentioned one to two times were included in the measure.

All items on the measure and representative quotes from part one participants endorsing the associated benefit are shown in Supplementary File A. Benefits clustered around four different types of benefits or “themes” (i.e., proposed factors). The first theme included benefits associated with life perspective and priorities. Participants spoke about how SB has helped them appreciate good moments, gain a sense of what’s important in life, fight for what’s right for others, be understanding of diversity and people of different backgrounds, look for the positive, live in the moment, accept and embrace who they are, be less self-conscious about their appearance, appreciate life, take full advantage of opportunities,¹ not sweat the small stuff, and have a stronger faith and spirituality. The second theme that emerged was personal characteristics and traits that adolescents and young adults with SB attributed to SB. Individual benefits included in this theme included a good sense of humor, being mentally strong and tough, being more sensitive, kind, and aware of how others are feeling, being open-minded and nonjudgmental, more self-aware, courageous, mature, and patient. The third theme was connection and opportunities and included opportunities to educate and inform others about SB, unique opportunities such as adaptive sports, closeness to family members, connection to others with SB and disabilities, enjoyment from helping others, and close relationships. Finally, a fourth type of benefit emerged around the concept of problem-solving abilities born out of experiences living with SB. These benefits included the ability to problem solve, learning to overcome difficulties, seeing that you can surpass other’s and your own expectations, and learning to adapt to difficult circumstances.

Based on the different benefits endorsed by participants during interviews, a measure with 31 items across four different categories (themes/factors) was piloted in part two. The last six participants in part one completed the same interview that the first 14 participants completed but were also shown the draft of the measure generated from earlier interviews and asked to provide feedback. None of this feedback suggested addition or removal of any items.

Part two methods

Participants

Participants in part two of the study, which aimed to validate and confirm the factor structure of the newly developed measure, were recruited from three sources: (a) ISBA, (b) Spina Bifida Texas, and (c) the national Spina Bifida Association (SBA). All recruitment partners advertised the study via their social media pages and/or emailed members of their association with our recruitment flyer. Interested participants reached out to study investigators via email. Eligible participants: (a) had SB, (b) were between the ages of 15 and 30 years, and (c) were able to answer the survey questions without the assistance of others (ensuring privacy and honesty in responses). Compensation was provided via a $25 Amazon electronic gift card.

Procedure

Part two participants were recruited between February and May 2022. Consistent with University Institutional Review Board (IRB) compliance and previous national online surveys conducted with individuals who have SB (Kritikos et al., 2021a), participants were consented via digital waiver of documented consent. For participants ages 15–17 years old, parents provided digital waiver of consent, and adolescent participants provided assent.

Measures administered in part two of study

Demographic and medical information, including age, gender, race and ethnicity, education, employment status, type of SB, shunt status, and lesion level, were collected via self-report questionnaire in order to characterize the sample.

The “Benefit-Finding and Growth Scale for Spina Bifida” (BFG-SB) developed from part one of the present study included 31 items, all beginning with “Spina bifida has…” and including a potential benefit. Each item is rated on a 5-point scale (1 = Strongly Disagree to 5 = Strongly Agree). The measure was scored by taking the mean of all the items (for total score) or mean of all the items in the subscale (factor scale score). Higher scores reflect higher levels of benefit-finding and growth.

“Optimism” was assessed with the 6-item Life Orientation Test-Revised (LOT-R; Scheier et al., 1994). An example item is “In uncertain times, I usually expect the best.” Participants rated the extent of their agreement with each item (0 = Strongly Disagree to 4 = Strongly Agree). Higher scores reflect higher levels of optimism. The measure has demonstrated good predictive and discriminant validity (Scheier et al., 1994); however, it demonstrated only moderate internal consistency in our present sample (α=0.66). Similarly low alphas (e.g., 0.48–0.74) have been found in clinical samples, possibly due to the low number of items in the scales (Hinz et al., 2022).

“Positive affect” was assessed using the positive scale of the Positive and Negative Affect Schedule—Short Form (PANAS-SF; Watson et al., 1988). Participants rated the extent to which they experience 20 different emotion words (e.g., interested, distressed, excited) on a 5-point scale (1 = Very Slightly or Not at all to 5 = Extremely). The positive scale is scored using responses to the 10 positive words on the measure, with higher scores reflecting greater positive affect. The positive affect scale demonstrated good reliability in the current sample (α=0.84).

“Life satisfaction” was measured with the Student’s Life Satisfaction Scale (SLSS; Huebner, 1991). The seven items on the scale include perceptions about one’s life and circumstances, e.g., “My life is going well” and “I would like to change many things in my life” (reverse-scored). Respondents rate each item on a 6-point scale (1 = Strongly Disagree to 6 = Strongly Agree). Higher total scores represent greater life satisfaction. The measure had acceptable internal reliability in the current sample (α=0.74).

Data analytic plan

Consistent with Holmbeck & Devine’s (2009) guidelines, we conducted factor analyses, evaluated reliability (internal consistency), and evaluated convergent validity for the measure. Confirmatory factor analyses (CFA) were conducted with Mplus Version 8 (Muthén & Muthén, 2017). Two CFA were compared, a one-factor model (all items loading onto one factor) and a four-factor model (life perspectives and priorities, personal characteristics and traits, connection and opportunities, and problem solving). A threshold of 0.400 was used to indicate a good factor loading. Estimated models were compared by examining various fit indices including the comparative fix index (CFI; ideal study criterion ≥0.95), Tucker–Lewis index (TLI; ideal study criterion ≥0.95), and root mean square error of approximation (RMSEA; ideal study criterion ≤0.05; Hu & Bentler, 1999; Kline, 2023). A chi-square difference test was used to determine whether a more constrained model (one-factor CFA) resulted in a significant deterioration in model fit over the less restrictive model (e.g., four-factor CFA), with a significant test indicating that the less restrictive model was better fitting (Cheung & Rensvold, 2002). In addition to formal statistical testing, the overall global model fit (i.e., highest CFI and TLI indices; lowest RMSEA indices) was assessed.

Descriptive and correlation analyses were conducted in IBM SPSS Statistics (Version 29.0). Pearson r correlations were used to examine the associations between the benefit-finding and growth measure and three measures chosen to assess concurrent validity. Data are available on request.

Part two results

Sample characteristics

Part two included 251 adolescents and young adults with SB. The sample had a mean age of 22.86 years (SD = 3.35; Mdn = 23.0; range: 15–30 years) and had a fairly even gender representation. Participants self-reported their racial and ethnic identity, resulting in some participants identifying with only one identity (e.g., White without specification of ethnicity, or Hispanic/Latine without specifying a racial identity). The most commonly represented group was White without an ethnicity selection, followed by African American without an ethnicity selection. The most frequently reported highest level of education was “finished high school,” and the most common employment status was part-time employment. Nearly half of the participants endorsed a history of mental health care treatment. Table 2 provides additional descriptive information about this sample. Of note, the shunt rate in this sample (34.3%) is particularly low when compared to the SB population (Copp et al., 2015).

Table 2.

Demographic and medical characteristics of 251 survey participants.

Characteristic	N (%)
Age, M (SD; range)	22.86 (3.35; 15–30)
Gender
Male	121 (48.2%)
Female	130 (51.8%)
Race/ethnicity
African American	54 (21.5%)
Asian	11 (4.4%)
White	142 (56.6%)
Latine	32 (12.7%)
Multiracial	10 (4.0%)
Did not identify	2 (<1%)
Highest level of education
Finished grade school	5 (2.0%)
Some high school	38 (15.1%)
Finished high school	63 (25.1%)
Business or technical school	22 (8.8%)
Some college	61 (24.3%)
Finished college	44 (17.5%)
Attended graduate or professional school	16 (6.4%)
Received a professional degree	2 (0.8%)
Employment status
Unemployed	71 (28.3%)
Volunteer	3 (1.2%)
Student	45 (17.9%)
Disability from work	13 (5.2%)
Employed, part time	71 (28.3%)
Employed, full time	47 (18.7%)
Missing	1 (0.4%)
Marital status
Married	17 (6.8%)
Single	201 (80.1%)
Living with significant other, not married	24 (9.6%)
Separated	4 (1.6%)
Missing	5 (2.0%)
SB severity composite, M (SD; range)	1.67 (0.38; 1–2.50)
SB type
Myelomeningocele	77 (30.7%)
Meningocele	59 (23.5%)
Lipomeningocele	16 (6.4%)
Lipmyelomeningocele	58 (23.1%)
Lipoma	36 (14.3%)
Unsure	4 (1.6%)
Missing	1 (0.4%)
Timing of SB diagnosis
During mother’s pregnancy	137 (54.6%)
After birth	106 (42.2%)
Unsure	7 (2.8%)
Missing	1 (0.4%)
Shunt status
Yes	86 (34.3%)
No	147 (58.6%)
Unsure	14 (5.6%)
Missing	4 (1.6%)
Shut revision
Yes	35 (40.7%)
No	45 (52.3%)
Unsure	5 (5.8%)
Missing	1 (1.2%)
Number of revisions, M (SD; range)	4.56 (4.84; 1–20)
Lesion level
Sacral	50 (19.9%)
Lumbar	162 (64.5%)
Thoracic	12 (4.8%)
Unsure	24 (9.6%)
Missing	3 (1.2%)
Toileting
With help	110 (43.8%)
Catheterization, daily regimen	155 (61.8%)
Participation in bowel program	112 (44.6%)
Diaper use	80 (31.9%)
Use of
Orthotic braces	85 (33.9%)
Crutches	82 (32.7%)
Wheelchair	143 (57.0%)
Walker	47 (18.7%)
Ever received mental health services
No	132 (52.6%)
Yes	118 (47.0%)
Missing	1 (0.4%)

Factor structure of BFG-SB

Model fit statistics can be found in Table 3. The one and four factor models had the same RMSEA, CFI, and TLI scores, which indicated excellent fit (RMSEA=0.06, CFI=0.96, TLI=0.96). Although the four-factor model was significantly better fitting based on the chi-square difference test, since the fit indices were the same across models, the rule of parsimony suggests that we should conclude that the best model is the one-factor model (Alavi et al., 2020). The four-factor model is still appropriate based on the model fit statistics, however. The standardized factor loadings for the four-factor model are presented in Table 4. For the four-factor model, all items loaded above 0.40 and loaded significantly on each factor. Higher standardized loadings are evidence of convergent construct validity (i.e., the item measures the factor it was intended to measure).

Table 3.

Model statistics for BFG confirmatory factor analyses.

	RMSEA	CFI	TLI	χ2 difference test	df	p-value
AYA-SB reported benefit finding
One-factor	0.05	0.96	0.96
Four-factor	0.05	0.96	0.96
One-factor vs four-factor				16.69	6	.0106

Note. Bolded row represents the best fitting model. RMSEA = root mean square error of approximation, CFI = comparative fit index, TLI = Tucker Lewis index, df = degrees of freedom.

Table 4.

Factor loadings for four-factor BFG model.

Items	BFG AYA-SB
Life perspective & priorities
2. Spina bifida has helped me appreciate the good moments	0.61
4. Spina bifida has given me a good sense of what’s important in life	0.74
12. Spina bifida has taught me to fight for what’s right for people with spina bifida.	0.79
13. Spina bifida has taught me to fight for what’s right for people in general.	0.63
17. Spina bifida has helped me be understanding of diversity and people of different backgrounds.	0.67
18. Spina bifida has taught me to look for the positive in situations.	0.81
21. Spina bifida has taught me to live in the moment.	0.62
23. Spina bifida has taught me to accept and embrace who I am.	0.73
24. Spina bifida has made me less self-conscious about my appearance.	0.47
25. Spina bifida has helped me appreciate life.	0.64
27. Spina bifida has helped me to take full advantage of life’s opportunities.	0.69
30. Spina bifida has taught me not to sweat the small stuff.	0.59
31. Spina bifida has strengthened my faith and spirituality.	0.51
Personal characteristics and traits
6. Spina bifida has given me a good sense of humor.	0.48
7. Spina bifida has made me mentally strong and tough.	0.65
10. Spina bifida has made me more sensitive, kind, and aware of how others are feeling.	0.69
16. Spina bifida has made me open-minded and nonjudgmental.	0.71
19. Spina bifida has made me more self-aware.	0.71
26. Spina bifida has made me courageous.	0.71
28. Spina bifida has made me more mature.	0.74
29. Spina bifida has made me patient.	0.63
Connections and opportunities
3. Spina bifida has given me the chance to educate and inform others about spina bifida.	0.71
8. Spina bifida has given me unique opportunities (for example, adaptive sports, camps, trips, special events).	0.53
9. Spina bifida has made me close to my family.	0.63
11. Spina bifida has connected me with the spina bifida community and others with disabilities.	0.69
20. Spina bifida has led me to enjoy helping others.	0.73
22. Spina bifida has led me to develop close relationships with others outside my family.	0.66
Problem solving
1. Spina bifida has taught me to problem solve.	0.54
5. Spina bifida has taught me to overcome difficulties.	0.72
14. Spina bifida has shown me that I can surpass other people’s and my own expectations.	0.77
15. Spina bifida has taught me to adapt to difficult circumstances.	0.78

Descriptive data on scores/reliability of BFG-SB

Model fit statistics supported the use of both a total score (one-factor model) and four subscales (four-factor model). The four subscales included (a) Life Perspectives and Goals, (13 items; alpha = 0.88; M = 3.79, SD = 0.65), (b) Personal Characteristics (8 items; alpha = 0.83; M = 3.78, SD = 0.68), (c) Connections and Opportunities (6 items; alpha = 0.77; M = 3.83, SD = 0.70), and (d) Problem Solving, (4 items; alpha = 0.74; M = 3.85, SD = 0.71). The total score (n = 31 items) also had excellent internal consistency (α=0.947). The full range of responses (1–5) was endorsed on each item, and there was no evidence of significant kurtosis (>±1) or skewness (>±1) on any of the 31 items.

Concurrent validity of the BFG-SB

All results from correlation analyses are shown in Table 5. The BFG-SB measure was significantly and positively correlated with optimism, positive affect, and life satisfaction.

Table 5.

Concurrent validity between BFG-SB and associated variables.

Domains and variables of the life crises and personal growth model	BFG life perspective subscale	BFG personal characteristics subscale	BFG connections and opportunities subscale	BFG problem solving subscale	BFG total score
Optimism	0.36**	0.31**	0.32**	0.32**	0.36**
Positive affect	0.56**	0.49**	0.50**	0.51**	0.56**
Life satisfaction	0.21**	0.21**	0.13*	0.26**	0.22**

^*Correlation is significant at the 0.05 level (2-tailed).

^**Correlation is significant at the 0.01 level (2-tailed).

Discussion

This study aimed to develop a new self-report measure of benefit-finding and growth that would (a) amplify the voices of individuals with SB and (b) honor the strengths and positive lived experiences of these individuals. To achieve these aims, we collaborated with 20 adolescents and young adults through open-ended, qualitative interviews to generate items for the new measure. Six of these youth also provided feedback on the draft of the measure before it was piloted in a larger sample. Although this portion of our study was exploratory, we expected that adolescents and young adults with SB would endorse a range of benefits. Indeed, they described a wide range of benefits and possible items for the measure, many of which were not represented in previous measures of benefit-finding and growth, especially those available to pediatric populations (Benefit and Burden Scale for Children [Currier et al., 2009], Benefit-Finding Scale for Children [Phipps et al., 2007], and Posttraumatic Growth Inventory for Children [Kilmer et al., 2006]; see Kritikos et al. [2021b] systematic review for further detail). For example, spina bifida “has taught me to fight for what’s right for people in general,” “has helped me be understanding of diversity and people of different backgrounds,” “has helped me have a good sense of humor,” “has made me open-minded and nonjudgmental,” “has led me to enjoy helping others,” and “has shown me that I can surpass other people’s and my own expectations” are some of the many items on this new measure that have not been represented on other available instruments. As such, these items may capture unique benefits about the experience of living with SB. Further, these items could be relevant to other adolescent and young adult medical populations. Our approach of partnering with youth yielded a greater number of benefits than seen in other measures (i.e., 10 benefit-related items in each of the previously mentioned scales). The process of developing these other measures involved traditional methods of adapting adult measures for younger populations and using researcher-led item selection. By combining this approach with feedback from lived experience experts (i.e., adolescents and young adults with SB), our inclusive measure development approach may have given us a richer and more comprehensive picture of the lives of youth with SB.

Following review and refinement of our items by an expert panel of researchers, we piloted a 31-item measure in a national sample of adolescents and young adults with SB to confirm the factor structure and examine correlates of the new measure. Based on the items we developed from our collaborative approach, we hypothesized that the new measure would have a four-factor structure, with factors including life perspectives and priorities, personal characteristics and traits, connections and opportunities, and problem solving. Model fit results supported both a one-factor (total score) and four-factor structure, with the factors consistent with our hypotheses. This new measure showed strong internal consistency both in terms of the total score and factor scores, and performed better psychometrically (i.e., consistent factor structure) than did an existing measure of benefit-finding and growth (Tomich & Helgeson, 2004) administered to adults with SB (Kritikos et al., 2021a).

We also examined the concurrent validity of our new measure with three measures of constructs that are consistently positively associated with benefit-finding and growth: optimism, positive affect, and life satisfaction (Kritikos et al., 2021b). In terms of optimism, we hypothesized that individuals who self-reported higher levels of optimism would be more likely to identify positive experience from the challenges of living with SB. This relationship has been uncovered in other pediatric populations (Kritikos et al., 2021b) and was supported in the present study. Similarly, the BFG-SB was positively associated with greater positive affect and life satisfaction. Given that benefit-finding and growth involves the cognitive process of “making space” for positive experiences even amid difficult ones, it makes sense that those who have higher benefit-finding and growth scores are then reporting more positive affective experiences, as has been seen in prior literature (Helgeson et al., 2006). Greater life satisfaction among those who are higher in benefit-finding and growth is also likely due to an ability to integrate positivity into one’s experience of living with SB.

The present study followed measure development guidelines from Holmbeck & Devine (2009) and addressed several recommendations for improving research on benefit-finding and growth in the pediatric psychology literature (Kritikos et al., 2021b), including examining the construct in a more complex pediatric condition, using the voices of adolescents and young adults (as opposed to just their caregivers), and increasing the racial and ethnic diversity of previous samples (e.g., Kritikos et al., 2021b). In addition, this research confirmed the factor structure and psychometric properties of the measure, allowing for greater confidence in its clinical use (Holmbeck & Devine, 2009). We also followed Kritikos et al. (2021b) recommendations to use open-ended and qualitative methods when studying benefit-finding and growth in a novel population, and included people with lived experience working with the population (i.e., expert panel of SB researchers and clinicians).

Findings should also be interpreted considering several limitations. First, in study one, the sample was skewed with regards to representation, limiting its generalizability. Specifically, there were no African American participants and respondents were predominantly female. Additionally, the age of the sample was older (M = 25.55) and therefore the measure may emphasize the experiences of young adults more than adolescents. This skew in representation was more pronounced among the sample of six participants who were asked to provide feedback on a draft of the measure. Thus, we did not have the opportunity to learn how younger adolescent, male, or more racially diverse participants may have responded to the draft of the measure. In part two, the shunt rate was particularly low in our sample, suggesting that our sample is likely medically high functioning. It is possible that given the nature of our study where we required independent completion of a battery of questionnaires online, our sample may have had lower SB severity overall. Future research should aim to example this relationship in clinic samples and samples with greater condition severity. Due to funding limitations and the fact that our measure was considerably longer than other youth measures of BFG (Kritikos et al., 2021b), we also had fewer than the recommended 10 participants per questionnaire item for CFA (Comrey & Lee, 2013; Yong & Pearce, 2013). Additionally, in part two, we did not include measures of IQ or cognitive functioning, which likely has important implications for the cognitive task of deriving benefit from adversity. Future research should account for neurocognitive factors, especially in an SB population where cognitive limitations are common (Stiles-Shields et al., 2019). Furthermore, future research should assess participant comprehension and understanding of the items. Finally, use of all self-report measures raises issues around common method variance; future research should incorporate multi-informant approaches.

Consistent with best practices, pediatric psychologists are tasked with (a) accurately assessing the difficulties and strengths of people with chronic illnesses and medical conditions and (b) collaborating with patients to develop clinical tools and interventions that will be effective, accessible, and equitable. The present study shows promise in our efforts to arm clinicians with better tools to achieve the first task. The measure has good psychometric properties and could be used to produce a total benefit-finding and growth score (single factor) or examine components of benefit-finding and growth (four factors). We propose that our approach of partnering with adolescents and young adults in the creation of this new measure not only demonstrated respect for the lived experience of individuals with SB, but also produced a more effective measure, as evidenced by psychometric properties that were superior to those of a generic measure used in adults with SB (Kritikos et al., 2021). This approach to measure development can also serve as a useful model for the second task of collaborating with patients. Whenever possible, the patients who will receive an assessment or intervention should be involved in the creation and development of that product. Future research should pilot the measure in a clinic setting to assess for feasibility and utility of the measure. Should the measure perform well in this setting, it could be a useful way to better understand adolescents and young adults with SB and identify their strengths and even areas that may be augmented.

Supplementary material

Supplementary material is available online at Journal of Pediatric Psychology (https://academic.oup.com/jpepsy/).

Author contributions

Tessa K. Kritikos (Conceptualization [lead], Data curation [lead], Formal analysis [equal], Funding acquisition [lead], Investigation [lead], Methodology [lead], Project administration [lead], Writing—original draft [lead], Writing—review & editing [equal]), Zoe R. Smith (Formal analysis [equal], Software [equal], Writing—review & editing [equal]), Colleen Stiles-Shields (Conceptualization [supporting], Methodology [supporting], Resources [supporting], Supervision [supporting], Writing—review & editing [equal]), Olivia Clark (Formal analysis [supporting], Writing—review & editing [equal]), Adrien M. Winning (Formal analysis [supporting], Writing—review & editing [equal]), Alana Encalade (Data curation [supporting], Project administration [supporting], Writing—review & editing [supporting]), Millicent Hendrix (Data curation [supporting], Project administration [supporting], Writing—review & editing [supporting]), Vicki S. Helgeson (Conceptualization [supporting], Supervision [supporting], Writing—review & editing [equal]), and Grayson N. Holmbeck (Conceptualization [supporting], Data curation [supporting], Formal analysis [supporting], Funding acquisition [supporting], Investigation [supporting], Methodology [supporting], Project administration [supporting], Resources [lead], Supervision [lead], Writing—review & editing [equal])

Funding

This research was supported by a fellowship from the Society of Pediatric Psychology, the Drotar-Crawford Postdoctoral Research Fellowship, awarded to T.K.K., as well as a Research Support Grant from Loyola University Chicago, and by grants from the National Institute of Nursing Research and the Office of Behavioral and Social Sciences Research (R01 NR016235), National Institute of Child Health and Human Development (R01 HD048629), and the March of Dimes Birth Defects Foundation (12-FY13-271). C.S.-S. is supported by a fellowship from the National Institute of Mental Health (K08 MH125069).

Data availability

Data available on request.

Conflicts of interest: None declared.