Translation and preliminary validation of the Brazilian family resources scale in a sample of parents of children with congenital Zika virus syndrome

1 Introduction

Zika virus infection emerged as a global public health concern in 2015 and continues to have a lasting impact on those who were affected by the epidemic. Although infection was typically associated with mild illness, the 2015-2016 Zika outbreak in Brazil occurred concurrently with an unusual increase in the number of infants born with microcephaly. Exposure to Zika infection during pregnancy is associated with congenital microcephaly and a constellation of other serious birth defects affecting the central nervous system, called Congenital Zika Syndrome (CZS) [1, 2]. From 2015–2019, 3,332 confirmed cases of CZS were reported in Brazil with nearly 70% of these cases occurring in the northeast region of the country [3]. Most children with CZS have significant functional impairments and neurodevelopmental skill deficits, but variability within developmental profiles regarding the specific areas of functioning impacted and severity of impact is observed [4].

Zika virus impacts not only children’s development, but indirectly their caregivers’ mental health and the overall wellbeing of the family [5]. Accumulating evidence suggests that parents of children with CZS experience reduced quality of life, fatigue, increased stress, lower life satisfaction, and psychological difficulties [6–8]. For example, in a study based in Rio de Janeiro and Recife, mothers of children with CZS reported more stress, depressive symptoms, and anxiety compared to caregivers of children without microcephaly and developmental delays [9]. In another recent study, de Souza and colleagues assessed general mental health, positive and negative affect, fatigue, and life satisfaction in 86 parents of children ages 1–20 months with CZS (M age = 9.67 months) [6]. Findings showed that 18.6% of parents indicated a negative evaluation of their mental health, and nearly 7% indicated poor mental health and probable emotional disorder. In multivariate models, mental health was predicted by less positive and more negative affect, lower life satisfaction, and more fatigue.

Recognizing the complex needs of children and their families, published treatment guidelines specify a family-centered, individualized approach to providing services for children with CZS and highlight the necessity of identifying supports for caregivers [10, 11]. Hallmarks of family-centered care in services for children with disabilities include presenting families with developmental information, helping to facilitate adaptations in daily care and family routines, and engaging families in long-term planning to help establish and maintain positive developmental trajectories for children [12, 13]. Despite attention to the needs of vulnerable children and families in Brazil’s guiding principles for health care and established federal policies [14–17], two recent systematic reviews suggest that family-centered practices are not routinely implemented in early intervention services [18, 19].

Marini and colleagues [19] suggest that a barrier to the provision of family-centered early childhood intervention in Brazil is the paucity of scientific literature to help guide practice. Specifically, they argue that there are large conceptual and empirical gaps related to the provision of family-centered practices in Brazil that must be addressed to align service provision for children with disabilities with recommended approaches to early intervention. The ability to quantify levels of family capacity and need is foundational for practitioners to effectively provide empirically supported family-centered treatments. Similarly, an ongoing challenge for researchers seeking to address these gaps is a dearth of validated measurement tools that provide insight into the needs and experiences of families caring for children with disabilities. A critical first step in supporting children and families in the early intervention context is identifying the presence (or absence) of family resources in intrapersonal, interpersonal, and environmental domains that could help families to reduce stress and experience an increased capacity to support their children’s learning and developmental outcomes [20]. Although measures of family resources exist in the extant literature, none have been translated and validated in Brazilian Portuguese. Consequently, practitioners in Brazil do not have a comprehensive, standardized way of assessing family resources, including both family needs and strengths.

The most successful outcomes in early intervention are demonstrated by young children who receive services early in development and for a longer period, compared to older children receiving shorter amounts of services [21]. Thus, it is critical that early intervention services are engaging and impactful for families of young children with special needs, including those with CZS. Incorporating a focus on family resources is one way to promote family engagement that, in turn, increases achievement of treatment goals. Resource-based interventions involve identifying a family’s unique needs and using this information to help them acquire useful resources that promote both child and family wellbeing [22]. A first step in implementing tailored resource-based interventions for children with disabilities including CZS involves gathering accurate knowledge regarding the availability and adequacy of family resources. Although anecdotal evidence from Brazilian health care professionals suggests that inadequate family resources are a barrier to effective treatment, the availability of resources for families of children with CZS in Brazil has not been described adequately in part due to the lack of validated measures that are translated into Brazilian Portuguese and culturally adapted for this population [23].

2 Current study

The purpose of the present study was to advance the provision of family-centered early childhood intervention in Brazil by translating and providing preliminary validation of a questionnaire to help guide family-centered care-oriented professionals in developing effective family resource-based interventions. Simply translating an existing measure into a different language does not ensure cultural appropriateness or that the translated measure retains the same psychometric characteristics as the original version [24, 25]. This is because a measure that has been validated in a certain context is bound to the cultural nuances of the sample, as well as time. Additionally, there could be varying dialects of the translated language that could lead to errors in the transformations of the words that alter the semantics [26]. For example, Borsa and colleagues [27] argue that a translation into Brazilian Portuguese focused merely on linguistic equivalency may result in an overly complex translation that the target population would not understand, or an overly simplistic translation in which the content in the instrument is too condensed. Thus, a cultural adaptation process was engaged that, in addition to translation, included outside reviewers and an iterative process with members of the target population to ensure the validity of the translated measure. After the culturally-grounded translation process was completed, resulting data were tested through psychometric evaluation to further establish validity.

3 Methods and results

5 Discussion

The absence of a translated and validated instrument for measuring family resources in the context of early childhood intervention in Brazil is a significant barrier for research and effective service provision. The purpose of this study was to translate and provide initial validation of the FRS to create a Brazilian Portuguese version of the measure that has both empirical integrity and cultural relevance for Brazilian families caring for children with special health care and developmental needs. A rigorous translation process that emphasized linguistics as well as cultural appropriateness was utilized following published guidelines for the cross-cultural translation of questionnaires [26, 43]. Face validity, content validity, internal consistency reliability, and construct validity were considered throughout the translation and evaluation of the questionnaire.

This process resulted in the B-FRS, which was determined to be both theoretically related and reflective of the contextual intent of the developers of the FRS [28] and was deemed appropriate for use with a Brazilian Portuguese-speaking population. The B-FRS includes 27 of the 30 items that comprised the original FRS. Dropped items asked respondents to rate the adequacy of the following resources: 1) Heat for your house/apartment, 2) Childcare/day care for your child(ren), and 3) Money to buy special equipment/supplies for your child. Most participants selected the “not applicable” response option for these resources, suggesting that the items were confusing or had low content validity for the target population. Question 1 (heat) may have been deemed not applicable by the participants due to the tropical climate in which they live. Heat may rarely be needed or simply not viewed as a resource by Brazilians living in Recife because it is a humid, tropical coastal city [44].

The perceived non-applicability of childcare availability for caregivers in this sample may reflect that care for very young children with disabilities is often mother-centered [45]. Previous research on families and mothers of children with disabilities has found that many mothers in varying cultural contexts end or interrupt their career to take care of their children [46, 47]. This may be especially true in the Brazilian context, as evidenced in phenomenological research with Brazilian mothers of children with disabilities [48]. Rodrigues et al.’s [49] qualitative investigation of Brazilian caregivers of children with intellectual disabilities found a common theme among the mothers interviewed in that they recognized the child’s care as “complex and difficult to share with other people” (p. 418). Brazilian caregivers may feel other people, including childcare providers, are not as equipped to meet their child’s needs, especially given the young age of the children. Additionally, Rodrigues also found that mothers in their sample relied on sisters, parents, or cousins to help with their child’s care sporadically, so that formal, consistent childcare or daycare was not seen as necessary [49]. Since the caregivers in this sample reported having optimal adequacy regarding access to babysitting, it may be that caregivers were not relying on formalized, paid childcare and instead relying on family members and friends for more informal sources of help with their child as needed.

Money to buy special equipment or supplies for the child may not have been relevant in this context because the children were too young to need any specialized equipment. It is possible that caregivers could find specialized equipment like wheelchairs or glasses more important as their child ages. Alternatively, caregivers may not have been aware of any additional supplies or equipment their child might need at present or in the future. Other published studies reveal that parents of young children with CZS receive little guidance, support, and education related to caring for their child, so it is possible they did not find this questionnaire item relevant to their experiences [50, 51]. Some caregivers have reported that health care workers and providers have sometimes been reluctant to help them with their child, so it is also possible that the caregivers were not made aware of any additional services from which their child could benefit [49, 52]. Additionally, the hospitals where the children were receiving services may not have been equipped with specialized materials themselves, so they may not have had any to give. Overall, however, examination of the remaining 27 items suggested that the B-FRS was sensitive to detecting differences among families in the adequacy of various resources, evidenced by ratings for most individual items spanning the possible range of response options.

Questionnaires that are adapted for use with samples that are culturally, linguistically, or otherwise different from the sample used in instrument development often do not retain the same psychometric characteristics as the original versions [25]. With respect to the FRS, mixed findings have been reported in every study exploring the psychometric qualities of the questionnaire, leading researchers to caution that the scale should be tested for validity and reliability when used in other settings and with other populations [30]. The small sample size used in field testing prohibited factor analysis of the B-FRS. To identify an appropriate strategy for scoring the B-FRS, the completed instrument was scored following each of the other published scoring conventions [28–33], and the obtained internal consistency estimates for the total scale and subscales were compared with those reported in prior studies.

The results revealed that the scoring approach identified by Patwardhan and colleagues [30] provided the best fit for the B-FRS. The aforementioned study was conducted in a sample of US families seeking assistance for their children with behavioral difficulties. Using exploratory factor analysis, the authors determined that a four-factor solution provided the best fit for their data, although this solution did not replicate any of the existing factor solutions found in prior psychometric investigations of the FRS. The same four-factor scoring approach was adopted for the current sample because it yielded internal consistency estimates for the four subscales that were the most acceptable, compared with other published scoring approaches. Moreover, the scoring rubric utilized in this approach [30] was the most robust in terms of dealing with missing data and using a mean scoring approach versus a sum score approach.

Comparison of the means and SDs for the four subscales in this sample and the Patwardhan sample revealed that the current sample reported a lower average rating of resource availability for three of the four subscales as follows: Basic Needs, 3.42 versus 4.45; Time for Family, 2.72 versus 3.93; and Essential Care, 2.85 versus 4.01. These results indicated that family resources were much lower for Brazilian families compared with Patwardhan and colleagues’ American sample and were inadequate in this sample of families of children with CZS. This may have important implications for the effectiveness of interventions and long-term developmental outcomes for children with CZS in Brazil. Consistent with a hierarchy needs perspective, unmet basic needs take precedent in terms of influencing and directing human behavior [53, 54]. Consequently, families of children who report low basic needs may be more likely to devote their time and energy to trying to get these critical family needs met, versus spending time addressing children’s therapeutic needs, which may be perceived as an important but less immediate need for the family [28]. Further, to the extent that a family is expending time and energy into getting basic needs met, adding additional demands such as professionally-prescribed treatments may have the unintended consequence of increasing family stress, which in turn may worsen both child and parent health and wellbeing.

The findings revealed significant associations between low family resources and indices of parental psychosocial wellbeing. Specifically, the strongest relation was observed between basic needs and depressive symptoms. Considering that parents and caregivers of children with developmental delays and disabilities have been shown to be at an elevated risk for symptoms of stress and depression [55, 56], it was not surprising to see evidence of this in the sample. Having lower resources to meet the family’s basic needs was associated with higher depressive symptoms. Lower basic needs were also associated with lower levels of coping strategy usage. Since healthy coping strategy use in caregivers of children with disabilities has been shown to mitigate stress, families that have sufficient resources to meet their most basic needs may be buffered from experiencing negative mental health outcomes [57]. It may be helpful for service providers to consider whether a family is able to meet its most basic needs and, if not, address this in the child’s intervention or therapy. Ensuring that families have their basic needs met may improve the child’s therapy outcomes if it reduces caregiver stress and risk for mental health problems. Another strong, negative relationship was observed between extra time for family and depression and stress. This finding is consistent with a cross-sectional survey of mothers of children with disabilities in the US, in which perceived social support was found to be a protective factor between child behavior and maternal depression [58]. Family-centered interventions and therapies that build services into the family’s everyday lives, thereby giving the caregivers more time to spend with their family and less time isolated or traveling with their child, may be beneficial.

Overall, these results were broadly consistent with the social determinants theoretical framework which focuses on understanding how the circumstances in which people live shape their mental health [59]. This framework hypothesizes that populations with economic and social vulnerabilities (e.g., low socioeconomic status, unreliable employment, inadequate social support) are disproportionately affected by poor mental health and that cumulative stress and physical health are important factors through which the adverse impacts of social determinants intensify [60]. The construct of social determinants in relation to Brazilian caregivers’ mental health may encompass a wide range of factors, including societal influences such as economic opportunities, individual characteristics such as age and gender, and malleable factors such as educational attainment, financial strain, living conditions, health behaviors, and child disability and/or health-related needs [61].

Successfully addressing family resources and the social determinants that impact them in the context of early intervention will require changes to the ways that early intervention services in Brazil are conceptualized and delivered, as well as increased federal funding that can help to support programs seeking to align their service approach with best practices in family-centered early intervention. At present, children and families receive early intervention services primarily through the Sistema Único de Saúde (SUS), Brazil’s government-run public health care system. The SUS is the largest public health care system in the world and provides health care services free of charge to nearly 100% of the Brazilian population. However, despite clear recognition of the importance of early intervention for children with special needs in both the SUS principles and Brazil’s established federal health care policies (e.g., National Policy for Comprehensive Child Health Care, the National Health Policy for Persons with Disabilities and the Disability Health Care Network, and the National Primary Care Policy), existing laws in Brazil do not translate into all children in need receiving early intervention services because these policies are not adequately supported by federal funds. Thus, many children cannot access and benefit from intervention services. Moreover, there are indications that critical family resource needs are not being addressed, even among children and families who do receive services. Specifically, two recent systematic reviews suggest that family-centered care principles, which acknowledge the key role of family resources, are not routinely implemented in the early intervention approaches and specific practices used with young children who receive services [18, 19]. This may be because early intervention services are embedded within the SUS health sector and therefore guided by a medically-oriented rehabilitative care model. Intervention practices are aimed at stimulating skill development in specific areas of identified developmental deviation (i.e., motor, sensory, proprioceptive, speech-language, and social-emotional skills), with service providers including physical therapists, speech therapists, and psychologists. Ecological approaches that extend assessment and intervention into family and community contexts and consider influences such as the adequacy of family resources are not currently standard care in early intervention in Brazil. Rather, family support for children with CZS has been provided primarily via community-based programs that are not integrated into more formal government-funded early intervention services or through grassroots informal support networks that vary in structure and aims [18, 63].

6 Conclusions

This study has important implications for researchers and practitioners focusing on children with special health needs and their families. Having a reliable and valid measure of family resources can help researchers to more rigorously investigate how family resources influence service utilization patterns in the field of early intervention, modifiable factors related to treatment adherence, and the links between family resources and children’s ultimate developmental outcomes [32]. For early intervention service providers, the B-FRS appears to be a promising tool for assessing the adequacy of resources in families of children with complex developmental needs in Brazil. As an assessment measure, the B-FRS can be used to better understand the adequacy of a wide range of resources that are relevant to intervention planning and resources-based practices. For example, interventions may focus on helping families acquire resources to satisfy critical basic needs to improve intervention effectiveness. This is a crucial first step in exploring how family needs and resources can shape early intervention in a Brazilian context, promoting positive developmental trajectories for the child and family.