Language and Latino immigrants living with spina bifida: Social determinants of health – the missing dimension in quality of life research

In accordance with PRISMA guidelines, 1,184 articles were identified, of which eighteen US-based studies met the inclusion criteria for the systematic review (Fig. 1).

4. Discussion

Our findings document that individuals living with SB face many factors that negatively impact their reported QOL; these challenges, as well as the QOL instruments used to assess them, are wide-ranging. Along with physical domains, a number of additional factors were associated with lower HRQOL scores; the investigation and understanding of these factors as a basis to develop interventions designed to improve QOL emerged as an important pursuit. Consistent with previous reviews of HRQOL in patients with SB [26, 48], our findings demonstrated that most studies were cross-sectional in nature and described varying degrees of HRQOL divergence on parent and self-reported scores related to emotional, social, and school domains (Table 1). Moreover, a generic measure of QOL was utilized by the majority of studies ( n = 15/18; 83%), of which two were supplemented with the use of the SB-HRQOL [43, 44]. Although generic assessment tools allow comparisons of QOL among individuals with various chronic health conditions, their questions are not specific to the SB population. For instance, the physical health domain questions include items regarding “walking” and “running,” which are inapplicable to individuals who rely exclusively on a wheelchair for mobility. In contrast, SB-specific QOL instruments ask questions appropriate for individuals with limited mobility and address degrees of bladder and bowel incontinence that greatly impact an individual’s perceived QOL [46].

Thus, current research reasoning has evolved into calling for suitable QOL psychometric instruments that are condition-specific and clinically validated assessment tools that also value individuals’ perceptions of their HRQOL rather than unduly focusing on physical function [48, 49]. The SB-specific QOL instruments are more appropriate for individuals with SB because they offer insight into the person’s perspective on QOL. Instruments that increasingly incorporate the individual’s perception ask relevant questions related to self-esteem, independence, relationships, and bladder/bowel function, which are essential contributors to overall QOL [9]; in part, SB-specific QOL measures aim to quantify QOL through questions regarding embarrassment, lifestyle, and coping behaviors related to self-catheterization as well as incontinence. Conversely, generic QOL instruments fail to address concerns related to common SB symptoms and focus more on questions such as ambulation. Nonetheless, in our review, the generic PedsQL 4.0 questionnaire was found to be the most pervasive instrument used to measure QOL in individuals with SB. By contrast, only a minority of studies documented solely utilizing a SB-specific instrument for assessment [13, 45, 46].

The role that cultural context, as a SDH, plays in the outcomes related to chronic health conditions has long been identified [16, 50, 51, 52]. In particular, language plays a key role as a factor associated with outcomes among Hispanics/Latinos [53, 54]. Given the fundamental nature of language, we focused on it as a primary SDH. Health disparities research suggests that among some specific racial/ethnic groups, inferior health status may be explained partly by divergent SDH (e.g., social gradients, social exclusion, and changes in the health care system) [55]. It is also acknowledged that in order to properly address gaps in providing care for patients with SB, a national research strategy must be multi-centered, longitudinal, and inclusive of ethnic and socioeconomic diversity [56]. Promisingly, investigators have also begun to recognize existing disparities through the use of the National Spina Bifida Patient Registry (NSBPR) – a partnership between the CDC and SB clinics throughout the US. Schechter et al. [57], established that non-Hispanic blacks were less likely than the other groups to have documented bladder/bowel continence, as also were Hispanics/Latinos less likely than non-Hispanics. The authors went on to state that after controlling for SB-related intrinsic characteristics, significant variations in outcomes associated with age, gender, race/ethnicity, and insurance status remained. They demonstrated that non-Hispanic blacks, Hispanics/Latinos, and those without private insurance had less favorable outcomes. A second NSBPR study recognized that the likelihood of having bladder continence was significantly higher among non-Hispanic whites and those with private insurance [58]. A third NSBPR study demonstrated that non-Hispanic white race/ethnicity and private insurance were associated with an increased likelihood of receiving surgical services to treat neurogenic bowel (e.g., ACE procedure) [59].

In the face of these health disparities, the timely and proper study of QOL among the Hispanic/Latino population, which has the highest SB prevalence [1], is clear. Furthermore, it has long been recognized that measuring outcome indicators based on the differing cultural aspects of well-being – indicators such as QOL – is crucial [15]. In a previous study beyond health measures alone, Chowanadisai et al. [60] found that well-being assessment, such as satisfaction with health care, is significantly lower among Hispanics/Latinos with SB, even after controlling for socioeconomic status. Since the multidimensional conceptual understanding of an individual’s own QOL is thought to be profoundly impacted by the intrinsic cultural context [52, 63], health professionals are called upon to consider the impact of culture, ethnicity, and immigration status on QOL [61] in an increasingly ethnically diverse society. Additionally, even after adjustment for family income and parental education, language and culture are known to contribute to the disparities in health outcomes found among Hispanics/Latinos [64]. However, even though it has been demonstrated that culturally sensitive interventions improve QOL and outcomes among Hispanic/Latino children with chronic health conditions [65] and it has been further postulated that such an approach may benefit children with SB [60], efforts towards this goal are nascent in SB care [66].

Whereas researchers utilized supplementary assessment measures to investigate additional dimensions (e.g., stress, anxiety, depression, cognitive function) that theoretically could impact QOL [36, 41, 42, 45], in our US-based review where an estimated 55 million Hispanics/Latinos live, we found no study that probed cultural aspects (e.g., language, acculturation, immigration, etc.) as potential modifiers on QOL. In fact, within the largest minority group and the fastest growing US population segment [25], among whom nearly 75% of individuals speak Spanish at home [25], there is a dearth of research on HRQOL among those living with SB. Of the 18 QOL studies identified, only six (33%) reported including Spanish-speaking individuals. In these studies, a generic QOL measure was used, at times supplemented with an ad hoc translation of a condition-specific tool. Although some initial steps were taken to address culturally-appropriate care [42, 43, 44], further work is needed. Despite Congress’ previous efforts to safeguard adequate representation of ethnic and racial minorities in federally funded research, our review failed to identify an adequate culturally validated Spanish-language, SB-specific QOL instrument; this goal is especially relevant to parents and to new immigrants providing self-report. As we grow in our understanding of immigration as a SDH and its effects on health and well-being [67], this gap also becomes self-evident.

Of note, the literature reviewed showed no consistent expression to describe people whose ancestry could be traced to the Spanish-speaking regions of Latin America, including the Caribbean, Mexico, Central America, and South America. Moreover, in some instances, even the term “Spanish origin” was used [36]. Such terminology “places narrow and undue emphasis on the European influence of Spanish colonialism” [68]; thus, the root term Latino would have been preferable.

The authors acknowledge several limitations common to systematic reviews. Inherent limitations exist due to the nature of synthesizing large datasets. To reduce some of these limitations, such as selection bias and inclusion of irrelevant validity/reliability results, a very stringent search criteria was implemented by two reviewers who assessed all studies in accordance with PRISMA guidelines. A second limitation of this review may be that as language and culture were not primary outcomes for many of the QOL studies, they may not have reported all relevant information due to journal limitations or the perception of this information as irrelevant. It is possible that studies that did not report any demographic information did in fact include Hispanics/Latinos or had few Hispanics/Latinos in the community. To address this limitation, we provided county census data [29] relevant to each study site to provide a crude contextual proxy of the percentage of Hispanics/Latinos in the primary catchment area where the research was conducted (Table 1). A third limitation arises from the heterogeneity of the populations (e.g., spinal cord injury) included in the reviewed studies. However, to ensure that we appropriately represented individuals with SB, we did not include any studies with relatively few participants with SB (i.e. studies with less than six participants with SB).

Since its inception [69], those evaluating QOL among individuals with SB have done a remarkable job in developing and moving forward this effort and its associated body of literature. However, these gains have not translated equally to all segments of the SB population. As a starting place, we would encourage the SB research community to include Hispanics/Latinos in QOL studies and conduct interviews and questionnaires in their primary language. After this step is taken, other means will be needed to further delve into additional SDH, health beliefs, and health literacy. Future QOL research should not only take preferred language into account, but it should also explore other dimensions keenly relevant to immigrants such as levels of family support, acculturation, adverse childhood experiences, and resilience.

To our knowledge, this is the first study to summarize the US-based QOL literature under a SDH conceptual framework and to identify gaps in the implementation of these efforts among the population most prevalently affected by SB [1]. Recently, in March 2017, the Spina Bifida Association brought together content experts to update the SB-healthcare guidelines [70]. As these guidelines – comprising 25 areas of care delivery – are implemented, fresh momentum towards the employment of HRQOL measures is likely to ensue. Therefore, nationally we now have renewed guidance and have the opportunity to care for all individuals and parents as whole-persons as they transition across the lifespan [13], not only addressing the medical condition but also holistically approaching them in their language and cultural context.

5. Conclusions

“Blue marble health” was introduced in 2013 as a policy framework to illustrate trends in the geographic distribution of neglected tropical diseases affecting at-risk populations that live not only in low-income countries but also in pockets of the populace in wealthier nations [71]. Since that time, it has been noted that this framework may also have a role in illustrating the SB paradigm in a high-income nation such as the US [72]. Globally, since 2000 there has been a 49% increase in immigration [22], thus linguistically-congruous study of QOL has enlarging relevance not only locally, but also abroad. In the US, in spite of Congress’s NIH Revitalization Act of 1993, calling for efforts to ensure that minority groups are included in representative numbers in research studies and that Hispanics/ Latinos have the highest reported prevalence of SB, few HRQOL studies have documented inclusion of this population. According to the most recent US Census forecasts on immigration and birth, this demographic group will increase from 17.4% to 28.6% of the total population in the approaching decades; hence, future research should include cultural context as a component in the study of the HRQOL construct and support the utilization of validated Spanish-language measures for its investigation.