IntroductionSpina bifida (SB) is a congenital disorder caused by incomplete closure of the neural tube that is generally accompanied by a variety of disorders, such as hydrocephalus, cognitive difficulties, bladder and bowel incontinence, and ambulatory dysfunction [1]. The management of SB requires not only multidisciplinary collaboration but also individualized care from caregivers because its clinical manifestations vary widely, with severity primarily determined by the level of spinal cord lesions. Caregivers of children with SB often face a range of challenges, having to balance the demands of long-term medical care, such as urinary and fecal incontinence [2], while also focusing on their child’s education and future [3].
Previous studies have primarily focused on the negative impacts of rearing children with SB, such as parenting stress [4,5]. A meta-analysis showed that families of children with SB and other chronic illnesses face the highest parental stress levels, highlighting the need for interventions aimed at improving marital relationships, enhancing social support, and equipping parents with strategies to manage parenting stress [6]. Compared with families with typically developing children, families of children with SB report higher levels of parenting stress related to managing children’s behavior, reflecting the challenges they face in addressing complex caregiving demands [7]. Further research has shown differences in stress sources between parents of children with SB: Fathers experience greater stress when dealing with children’s maladaptive behaviors and insufficient social support, whereas mothers’ stress is more closely related to the medical characteristics and dysfunction of the children [8]. Age also emerges as a significant factor affecting maternal parenting stress because older mothers report higher stress levels [9,10]. Among the medical factors, Kanaheswari et al [11] identified clean intermittent catheterization as the only significant contributor to maternal parenting stress in families of children with SB. Moreover, ongoing issues such as limited mobility and bladder and bowel dysfunction in school-aged children also contribute to persistent stress among parents of children with SB [12].
In the process of coping with parenting stress among caregivers of children with SB, social support may play a buffering role. Studies have shown that higher satisfaction with social support is closely associated with greater positive emotional adjustment in mothers [13]. Greater support from the family is also strongly linked to better physical health in mothers [14]. Furthermore, greater levels of social support are strongly related to fewer behavioral problems in children [8,15,16]. Notably, Feldman et al [16] demonstrated that social support plays a crucial moderating role between parenting stress and child adaptation. The aforementioned studies demonstrate that social support plays a critical role in multiple aspects of rearing children with SB. Meanwhile, further research underscores that, in pediatric rehabilitation settings, the importance of social support for families of children with SB cannot be overlooked [17]. In addition to mitigating negative emotions, social support contributes to the positive adjustment of caregivers. A study focused on caregivers of children with developmental disorders suggested that social support facilitates cognitive adaptation, thereby promoting positive emotional changes in caregivers [18].
Although caregiving challenges are substantial in families of children with SB, the demands associated with caregiving roles may create opportunities for personal growth and positive adaptation. Benefit-finding (BF) refers to the recognition of positive life changes that emerge from the process of coping with difficult life events [19]. Research on BF often focuses on the patients themselves [20-22], but caregivers of patients also experience BF. An increasing number of studies have reported positive changes, such as BF in caregivers of children with chronic illnesses [23-25]. In a qualitative study involving nuclear family members of children with chronic food hypersensitivity, a hierarchical BF process model was constructed. The findings suggested that parental modeling and reinforcement facilitated the sharing of positive experiences among family members [25]. This process highlights how BF emerges within families including children with chronic illness as a dynamic mechanism, where shared positive experiences not only strengthen individuals but also contribute to the overall harmony of the family unit.
In the context of SB, a study focused on young adults with SB found that the most prominent aspects of BF they experienced were related to personal strengths, interpersonal relationships, and life philosophy [26]. In addition, Kritikos et al [27] developed a BF scale specifically for individuals with SB based on a sample of 20 adolescents and young adults with SB. Although caregivers of children with SB are likely to experience more challenges, their special caregiving roles may also foster opportunities for positive psychological adaptation. However, research on BF by parents of children with SB remains limited, and the experiences of these families in different cultural contexts, such as Japan, have yet to be fully explored. Whereas the study by Kojo and Fukumaru [28] found that parents of children with SB tend to report high levels of depression, there remains a notable gap in understanding other characteristics, such as BF, parenting stress, and social support, of families of children with SB in the Japanese context.
In Japan, the existing caregivers’ BF scale was tailored specifically to caregivers of children with developmental disabilities [29], leaving a gap in measurement tools that adequately capture the BF experiences of caregivers of children with SB. The University of Washington Caregiver Benefit Scale (UW-CBS) developed by Amtmann et al [30] was designed to measure BF by caregivers regardless of whether their children have a medical condition. The flexible design of the UW-CBS makes it suitable for assessing BF in a variety of caregiving contexts.
Overall, this study sought to measure BF to explore the positive changes experienced by caregivers of children with SB. However, because no suitable scale exists in the Japanese context, the first objective of this research was to translate the UW-CBS into Japanese and evaluate its reliability and validity within the Japanese context. As little research in the Japanese context has focused on parenting stress, BF, and social support among caregivers of children with SB, the second goal of this study was to examine the current status of parenting stress, social support, and BF of caregivers of children with SB and clarify the unique characteristics of families of children with SB by including parents of able-bodied children as a comparison group.
DiscussionStudy 1The aim of Study 1 was to translate the UW-CBS into Japanese and validate its reliability and validity through a pretest and main survey. Because the UW-CBS was not developed in an Asian context, cognitive interviews were conducted as a critical step to address potential cultural differences in the process of scale adaptation. Overall, nearly all caregivers were able to understand the meaning of all items and found most of them to be clearly expressed. However, caregivers also experienced some hesitation and confusion while responding to certain items. Similar feedback was also provided when the UW-CBS was translated into European languages [34], suggesting that, although the scale is broadly applicable, some conceptual nuances require careful adaptation.
Regarding reliability, internal consistency was high (Cronbach α=0.92), but test-retest reliability yielded an ICC of 0.62 (P=.051), which did not reach the level of statistical significance. This marginal result likely reflects a lack of statistical power due to the small sample size (n=7) rather than the inherent instability of the scale. However, given that the original version demonstrated robust stability and our qualitative findings confirmed the items’ conceptual clarity, these results provide a preliminary indication of the scale’s reliability.
In adapting the UW-CBS to Europe, the mean score (44.50) was lower than that in the United States (47.90) [30]. In Japan, this study yielded an even lower mean score (43.20). In addition to the issues caregivers encountered while responding, this phenomenon may also be attributed to the cultural differences between Asian and Western contexts. Compared with those in the United States, Japanese adults report higher levels of pessimism and are less positive [44]. In addition, adults in the United States perceive self-esteem as both desirable and consequential, whereas Japanese people view self-esteem as desirable but not consequential [45]. Japanese respondents tend to use rating scales more moderately [46], which may also lead to lower observed mean scores. These differences in cultural perspectives may partially explain why the UW-CBS scores observed in this study were lower in the Japanese context, as a more pessimistic outlook and less emphasis on self-esteem may reduce the likelihood of identifying benefits in caregiving situations. In addition, interviews with caregivers about their BF experiences while rearing children with SB revealed several recurring themes, including psychological and emotional growth, improved knowledge and skills, expanded social support networks, changes in perspective, and planning for the future. The results of the semistructured interviews on BF indicated that experiences related to BF exist within caregivers of children with SB, and the themes appear to align with the content of most items on the UW-CBS, suggesting a potential connection between the scale’s construction and the lived experiences of the target population.
In the main survey, no ceiling nor floor effects were observed for the 13 items on the UW-CBS, and the total score followed a normal distribution. The absence of meaningful DIF across gender and age, indicated by McFadden R2 values <0.012, suggests that the UW-CBS demonstrates measurement stability. In addition, the overall Cronbach α coefficient for the Japanese-translated UW-CBS was 0.92. Whereas the original study reported that this correlation was lower than anticipated, the result obtained in this study was similar to the original result. A retest with 7 caregivers was conducted, and the test-retest reliability was 0.62, lower than that in the original study (0.92). Although the result in this study was not significant, likely because of the small sample size, the ICC of 0.62 still suggests that the UW-CBS demonstrates a certain degree of reliability. Construct validity was examined by exploring the correlation between the resilience scale and the UW-CBS. In this study, the correlation was r=0.30 (P=.02), which is comparable to the original study’s correlation of r=0.28 (P<.01). The known-groups validity comparisons identified differences in BF between caregivers of children with SB who differed in medical condition, as indicated by ventriculoperitoneal shunt use, as well as between caregivers with and without a partner, in the expected directions. These findings suggest that the UW-CBS is capable of capturing meaningful variation in BF across caregiver subgroups.
Study 2The aim of Study 2 was to explore the characteristics of caregivers of children with SB. Because little research has explored BF among caregivers of children with SB, we first examined the characteristics of their scores on the UW-CBS. We found that parents rearing only children with SB scored lower on the UW-CBS than did those who were also rearing other children without serious conditions. This result suggests that a greater number of children may offer caregivers increased emotional support or family resources, which may foster more positive emotional experiences. Previous research concerning parents of children with autism spectrum disorder has indicated that parents and siblings themselves believe that they mature more quickly as a result of taking on a caregiver role [47]. Furthermore, in a study of resilience among families of children with autism, interviewers also noted that siblings tend to look after the child with autism, thereby bringing family members closer together [48]. These findings may also be applicable to families rearing children with SB, where the presence of additional children may contribute to the development of a stronger support network among family members, thereby influencing caregivers’ perceptions of BF.
In this study, caregivers with partners had higher UW-CBS scores. This result aligns with previous research emphasizing the importance of marriage, as marital satisfaction contributes to improved psychological adjustment [6] and positive child adjustment outcomes [49]. Another study highlighted that a partner’s companionship is an indispensable source for mothers of children with SB, which cannot be easily substituted by other forms of social support [50]. By contrast, single mothers of children with SB reported significantly higher parenting stress [7,11], suggesting that the absence of a partner may exacerbate parenting challenges. Further research should place greater emphasis on providing support for single-parent families of children with SB.
The comparison between caregivers of children with SB and parents of able-bodied children resulted in a significant difference in social support scores, but no significant differences were observed in parenting stress nor BF. The significant difference in social support may stem from the extensive medical needs of children with SB, which often require ongoing help from health care professionals and social workers. In addition, caring for a child with SB can lead caregivers to connect with others in similar situations, further expanding their support networks. These assumptions are partially supported by the pretest findings, in which the category Expansion of social support networks emerged. Caregivers mentioned having access to help from specialists in areas like rehabilitation and welfare during difficult times, as well as forming friendships with other caregivers of children with SB.
In prior studies, caregivers of children with SB reporting higher levels of social support also tended to report lower parenting stress [8,16] and distress [13]. Despite the fact that parents of children with SB have generally reported higher levels of parenting stress in most studies [6-12], greater social support may account for the finding in this study that caregivers of children with SB experience levels of parenting stress comparable to those of parents rearing able-bodied children.
Although the original UW-CBS paper reported higher BF scores in community samples compared with those in medical condition samples [30], in this study, no significant difference in UW-CBS scores was found between caregivers of children with SB and the control group. This result may be attributed to the relatively high levels of social support received by the caregivers of children with SB in this study. A study focusing on caregivers of children with developmental disabilities explored a BF-related model, identifying a positive relationship between social support and BF while suggesting that social support nourishes framework reconstruction and potentially facilitates the BF process [18]. This finding raises the possibility that caregivers of children with SB, as fellow caregivers of children with chronic conditions, might experience a greater degree of BF in the context of a well-developed social support network.
LimitationsThis study has some limitations. First, the recruitment of caregivers was inherently challenging because of the medical complexity and care demands associated with SB, and recruitment was further constrained by the COVID-19 pandemic. As a result, the sample sizes for the main survey and retest were relatively small, which limited the statistical power. In particular, test-retest reliability did not achieve statistical significance, suggesting that the results regarding the scale’s stability should be interpreted as preliminary. Second, the validation of the scale was restricted to caregivers of children with SB as the target group. In future research, the application of the UW-CBS is anticipated to be examined in larger sample sizes and among a broader range of caregivers of children with special needs to evaluate the stability and measurement properties of the UW-CBS. Second, because this study had a cross-sectional design and measured BF at a specific point in time, longitudinal research should be considered to gain a deeper understanding of the positive psychological adaptation process of parents of children with SB within the Japanese context. Third, the recruitment strategy, which relied on associations and snowball sampling, may have led to an overrepresentation of caregivers with higher levels of social support. This may reduce the representativeness of the study sample. Future studies should adopt broader and more systematic recruitment strategies to enhance representativeness and reduce potential selection bias.
ConclusionsThis study, based on the context of Japanese caregivers of children with SB, translated and validated the UW-CBS, demonstrating its usability. In addition, BF was observed in families of children with SB, with more pronounced levels in families simultaneously rearing typically developing children and in households with a spouse or partner. Families of children with SB also reported receiving greater levels of social support than parents of able-bodied children.