Parental Health Literacy and Patient Clinical Outcomes in Juvenile Idiopathic Arthritis with a Health Equity Lens

1. Introduction

Juvenile idiopathic arthritis (JIA) is the most common primary rheumatologic condition that affects children, with a childhood arthritis prevalence of 305 per 100,000 US children and adolescents¹. JIA outcomes have substantially improved since the introduction of biologic disease modifying anti rheumatic drugs (bDMARDs)². However, significant gaps remain in understanding how patient outcomes vary among racial and ethnic backgrounds. A cross-sectional study of children with JIA enrolled in the Childhood Arthritis and Rheumatology Research Alliance (CARRA) registry found that African American children with polyarticular-course had higher risk of joint damage compared to white children, and that Hispanic children had increased odds of having disability scores higher than 75^th percentile compared to non-Hispanic/Latino children³. Similarly, Hispanic American patients had significantly increased joint disease activity, severity, and disability compared to non-Hispanic American patients in a large cohort ⁴.

In addition to ethnicity and race, social determinants of health influence clinical outcomes of patients with JIA. Another CARRA study demonstrated that lower household income was associated with higher disease activity, and that public insurance and lower family education levels were associated with persistent functional disability⁵. Although some of the differences in outcomes and prognosis may be explained by the JIA subtype and the presence of certain biologic markers, such as higher prevalence of rheumatoid factor and polyarticular involvement in Hispanic American patients⁴, studying how non-medical drivers influence the outcomes of patients with JIA is crucial to improve understanding and prioritize interventions.

Health literacy (HL) is an important non-medical driver of health which includes personal competencies and resources that are needed for individuals to access, understand, and use health information and services to make decisions about their health^6,7. Adequate health literacy has been associated with greater health knowledge in studies of child health^8,9. However, the effects of HL on health outcomes have been mixed. For example, a systematic review on HL and diabetes found that adequate HL was associated with appropriate diabetes knowledge, but a consistent relationship between HL and diabetes outcomes was not seen⁹. Individual studies have reported a relationship between higher HL and improved glycemic control¹⁰, as well as a relationship between limited HL and worse child development¹¹. Factors that influence limited HL in caregivers include foreign origin, limited educational attainment, younger caregiver age, and public insurance¹².

In pediatric rheumatology, Dogan et al evaluated HL in a Turkish population with JIA using several HL tools. They found that approximately a quarter of the parents had inadequate or problematic HL, and an association with health status and quality of life was not seen ^{13 14}. In contrast, Lazaroff et al demonstrated transition readiness in young adults with rheumatologic conditions in Ohio. The cohort was characterized by having adequate HL (98%), being white (78.6%), and completion of some college or technical school (66.7%), and almost a quarter of their households earned 100-150K per year ¹⁵.

Due to the chronic nature of rheumatologic disorders, patients and their families interact with the healthcare system on an ongoing basis. Inadequate HL may represent a leading health disparity with potential areas of intervention. In this study, we explored the relationship between parental HL and child health outcomes in a diverse population of patients with JIA.

2. Materials and Methods

After IRB approval was obtained from Baylor College of Medicine, rheumatology clinic schedules from Texas Children Hospital medical center and satellite clinics were reviewed weekly to identify patients with JIA. To be considered for inclusion, patients had to be age 18 or younger, have a diagnosis of oligoarticular (4 or fewer joints) or polyarticular (five or more joints) subtype according to the International League of Associations for Rheumatology (ILAR) criteria, and be present with a parent. Only English and Spanish speaking parents were included. Patients with systemic, oligoarticular extended, psoriatic, and enthesitis related arthritis subtypes were excluded. Eligible families were contacted before or on the day of the rheumatology visit. Electronic or written consent was obtained.

SDH questionnaire were given to parents using the Protocol for Responding to and Assessing Patients’ Assets, Risks, and Experiences (PRAPARE) tool ^16,17. For race, responders could choose “Hispanic or Latino” as a “yes or no” question. Individuals were asked to select all races that apply from: Asian, Pacific Islander, White, Native Hawaiian, Black/African American, American Indian/Alaskan Native and “other”. Additional demographics information was obtained through a separate one-page survey that included age of parent, knowledge and use of Medicaid transportation, primary language spoken, and duration lived in the United States. HL score was obtained using The Newest Vital Sign (NVS), a six-question screening tool that tests numeracy and literacy using a nutrition label¹⁸. An NVS score of 0-1 suggests high probability of limited HL; a score of 2-3 indicates the possibility of limited HL, and a score of 4-6 as having adequate HL.

Chart review was completed with the following information: patient’s age, JIA ILAR subtype, time in clinical course from diagnosis (0-6 months, 12 months to 2 years, <2 years), time from symptom onset to first rheumatology visit in months, clinical disease activity score by cJADAS (at diagnosis, 6 months, 12 months, and at the most recent visit), treatment including steroid courses per year if any, maintenance medications, time from order of biologic and conventional DMARD to administration, number of emergency room visits in the last 6 months that were attributed to JIA, patient portal activation and utilization, documentation of noncompliance by rheumatology provider in the last 6 months from recruitment visit, no-show rates on electronic medical record (for all primary and subspecialty visits).

The primary outcome was disease activity based on cJADAS at the most recent visit. The cJADAS is a validated JIA outcome measure that includes three composite scores: the physician global assessment visual analog scale (VAS) of 0-10, parent/patient global assessment VAS of 0-10, and active joint count ranging from 0-10^19,20. Patient reported outcomes were measured using the Patient-Reported Outcomes Measurement Information System (PROMIS). The PROMIS mobility and upper extremity involve a set of questions that evaluate functional mobility and have been adapted for children^21,22 as well as a parent proxy version²³. The PROMIS tool used in our study included Pediatric Mobility-Short Form 7a for children aged 8 or older, and Parent Proxy Mobility Short Form 7a for children less than 8.

For oligoarticular patients, to be able to see a difference between limited (1.5), moderate (2.5) and high disease (4) activity in the cJADAS, we calculated needing 36 patients to have 80% power. In the polyarticular group, to be able to see a difference between limited (2.5), moderate (4.5), and high disease activity (8.5), at least 15 patients were needed to have 80% power. All baseline characteristics were summarized with descriptive statistics. Characteristics and clinical outcomes were compared in two HL groups, limited HL (NVS score of 0-1) and possible limited/adequate HL (score of 2-6) using Kruskal-Wallis test, Wilcoxon rank sum test, Chi-square test or Fisher's exact test. Logistic regression for the odds of active disease (cJADAS>1) was performed on multiple patient factors. All statistical analyses were performed using Stata v 18.1 (StataCorp, College Station, TX, USA).

3. Results

A total of 75 parents were recruited for the study of which 15 (20%) had limited HL (NVS 0-1), 11 (14.7%) had possible limited HL (NVS 2-3), and 49 (65.3%) had adequate HL (NVS 4-6). Table 1 summarizes the baseline demographics of our study population. Median age of patients was 10 (IQR 7, 14), 36 (48%) had oligoarticular and 39 (52%) polyarticular subtype, and most patients had been diagnosed at least 2 years prior (72%). Among parents, most were white (64%), about half reported Hispanic ethnicity (47%), and 22 (29.3%) were Spanish speaking. Most patients had an active patient portal (98.7%). The median disease activity score was 2 (IQR 0,6) with most patients having active disease at the most recent visit (52.2%).

Parental age, patient age, and time since diagnosis were similarly distributed among groups. Possible limited/adequate HL was significantly associated with English language, white race, living in the United States for 10 years or more, higher education, private insurance, higher income, and seeing a loved one 5 or more times per week. The limited HL group had significantly higher percentage of Spanish speakers, Hispanic ethnicity, less than high school education, and no insurance. There was a tendency for patients with limited HL to select “I choose not to answer this question” in several of the PRAPARE questions including race (20%), “worried about losing housing” (13%), “knowledge about Medicaid transportation” (40%), and specific needs (food, clothing, utilities, childcare, healthcare, phone) (33.3%, P<0.023). There was no difference in household size, homelessness, worry about losing housing, use of Medicaid transportation, specific needs and frequency of feeling anxious or stressed (data not shown). When comparing the time of conventional DMARD vs biologic therapy initiation by HL group there was no significant difference.

Table 2 summarizes outcomes by HL group. Polyarticular and oligoarticular groups were evaluated separately for outcomes that were influenced by joint count. For polyarticular patients, median cJADAS was significantly higher in patients whose parents had limited HL [median cJADAS 8 (3,11) vs 2.2 (0, 7), p=0.017]. Only 1 patient (10%) in the limited HL group had inactive disease vs 13 (46.4%) in possible limited/adequate HL group, although not significant (p=0.059). There was no significant difference in mobility scores, number of steroid courses, or joint count at presentation, at 6 months or 12 months from diagnosis. For oligoarticular patients, median cJADAS was higher in patients in the limited HL group but the difference was not statistically significant. Combined patient and parental proxy mobility scores were significantly lower in oligoarticular patients in the limited HL group when compared to the possibly limited/adequate HL group [35.3 (27.1,45.4) vs 52.1 (52.1,52.9), p=0.037]. Similarly to polyarticular patients, only 1 patient (25%) in the limited HL group vs 18 (67%) in the possible limited/adequate HL group had inactive disease, but the difference was not statistically significant (p=0.272). There was no difference in the number of steroid courses or joint count at different time points in oligoarticular patients. Patients in the limited HL group had significantly higher no-show rates when compared to possible limited/adequate HL group [6 (2,12) vs 1 (0,4), p=0.006]. There was no difference in the time to diagnosis (data not shown), rates of recent ED visits or medication nonadherence among HL groups.

Table 3 summarizes demographics and outcomes by language. There was a disproportionate number of polyarticular patients in the Spanish group (72.7% vs 43.4%, p=0.025), as well as significantly higher percentage of patients with limited HL (p<0.001). Similarly to possible limited/adequate HL, English language was associated with living in the United States for more than 10 years (p<0.001), more than a high school education (p<0.001), significantly higher income (p<0.001) and having private insurance (p<0.001). When comparing region of heritage, Spanish speakers had higher percentage of Central American ancestry when compared to English speakers (50% vs 13.3%, p=0.023). There was no difference in age of patient, time in clinical course, successful use of Medicaid transportation, household size, and housing situation between groups. Spanish speaking patients had significantly higher cJADAS scores [3.5 (0.5,10.2) vs 1 (0, 5) p=0.033]. There was no difference in active vs inactive disease, number of ED visits, patient portal use, noncompliance, mobility scores, no-show rate, number of steroid courses, or joint count at different time points between language groups.

Table 4 depicts the results of a multivariable logistic regression for active disease based on cJADAS. After adjusting for insurance, language, compliance, JIA subtype, and ethnicity, limited literacy had higher odds of active disease (OR=6.85, [CI 1.18-39.58], p=0.032). .

4. Discussion

To our knowledge this is the first study evaluating HL using a validated tool in parents of patients with JIA in a diverse population. In our study, 20% of parents had limited HL, which is similar to that reported by Dogan et al¹³, but higher than the study performed by Lazaroff et al in which all parents had adequate HL¹⁵. Other studies that evaluate HL in patients with rheumatologic conditions report limited HL rates ranging from 7% to 42% ^24–26. Several factors may explain the prevalence of limited HL in our study population. Notably, the large proportion of immigrants may reflect limited familiarity with the U.S. healthcare system. Supporting this hypothesis, 80% of patients with limited HL reported Hispanic ethnicity, and over 30% had resided in the U.S. for less than years.

In our study, limited HL was associated with significantly higher disease activity in polyarticular patients and with an overall higher odds ratio of active disease. Although the difference in disease activity among patients with oligoarticular disease did not reach statistically significance, both the median disease activity score and the proportion of patients with active disease were higher in the limited HL group. The absence of a statistically significant association is likely attributable to the small number of oligoarticular patients with limited HL in our cohort, which included only 4 individuals. Additionally, oligoarticular patients in the limited HL group had significantly lower PROMIS mobility scores. Thus, our study supports that limited parental HL negatively affects clinical outcomes.

Although no association was seen between parental HL and JIA outcomes in the study performed by Dogan et al.¹³ and Lazaroff et al.¹⁵, these represented homogeneous populations with similarities in language and acculturation. Studies that have evaluated the role of HL in outcomes of rheumatologic conditions in diverse populations have also identified an association between HL and outcomes^26,27. A systematic review of 49 studies evaluated the role of HL and outcomes of children with chronic disease, including one study in patients with JIA, found that in both high income countries and low income countries, limited parental HL was associated with poorer child health outcomes²⁷. Higher HL in patients with childhood lupus has been associated with higher medication adherence and higher quality of life²⁶. A study that evaluated disease activity over time in 108 patients with rheumatoid arthritis found that patients with adequate HL had significantly lower disease activity over time than patients with several HL limitations²⁸.

In our cohort, the no-show rate was significantly higher in the limited HL group. Interestingly, when no-show rates were analyzed by language, no significant differences were observed, suggesting that health literacy (HL) may be an independent factor contributing to the higher no-show rates. Furthermore, there were no significant differences in transportation-related barriers between the limited HL group and the possible/adequate HL group. This finding indicates that transportation challenges are unlikely to explain the observed disparity in no-show rates. Instead, other factors may play a role, including confusion about appointment dates and times, limited access to patient portals and automated appointment reminders, inadequate understanding of the importance of regular follow-up visits, or difficulties obtaining time off from work to attend appointments. Patient portal use was also significantly lower in the limited HL group. A systematic review evaluating the effect of patient portals on health outcomes demonstrated improved monitoring of health status, physician-patient interactions and quality of care²⁹. Sun et al. examined the impact of several socioeconomic factors on patient portal utilization and found that individuals who identified as Black or African American, spoke Spanish as their primary language, were male, reside in rural areas, or lack health insurance had significantly lower odds of using patient portal³⁰.

Our cohort highlights the presence of multiple social, demographic, and environmental disparities that may influence HL. Parents with higher HL and English as their primary language have significantly higher income, education, private insurance, and are less likely to report specific needs such as food, clothing, utilities, childcare, healthcare, and phone. These differences stress the importance of getting social work involved in patients whose parents are Spanish speakers and who have limited HL to help address some of these barriers. One key difference between HL groups was the contact with a support system, with parents with limited HL having significantly less social support. This finding may be related to recent immigrant status among families with limited HL, as they may not had the opportunity to establish a robust social support network.

There was no significant difference in time to conventional DMARD or biologic therapy initiation between limited and possible limited/adequate HL. Access to medications may be related to Medicaid insurance coverage, specialty pharmacy services and patient assistance programs. A key program in our institution is the specialty pharmacy which delivers medications to patient’s homes, a strategy that is likely filling gaps in HL. It is worth noting, however, that both specialty pharmacies and patient assistance programs require significant interactions with the healthcare system such as providing proof of income and coordinating deliveries. Our institution helps overcome some of these barriers by having a high percentage of Spanish speaking staff (nurses, PA specialists, and physicians).

Research examining targeted health literacy interventions has shown improvements in health knowledge, health-related behaviors, and healthcare utilization^8,31,32. In a 2022 study of 13 young adult patients with systemic lupus who had successfully transitioned into adult care, patients expressed frustration and hesitance with doctor’s explanations regarding the disease process and the rationale behind treatments³³. The authors further noted physician’s explanations frequently relied on “doctor talk”, which may hinder patient understanding, and recommended cultural competency training to enhance physician-patient communication. Topics that patient expressed they personally felt they had difficulty with included how to identify doctors through insurance, difficulty with getting appointments, knowing how to ask for refills, and knowing their own personal medical history when asked at the adult visit. A systemic review of 31 studies evaluating the efficacy of educational interventions in adults with rheumatoid arthritis found significant effects on disability scores, joint counts, patient global assessment, psychological status, and depression³⁴. Studies evaluating the impact of educational interventions in JIA specifically are limited, but similar interventions can result in positive outcomes.

Limitations of our study include a small sample size. We think likely the HL rates are underrepresented given that patients with limited HL may have difficulty making it to appointments. Higher HL has been associated with higher research participation and limited HL has been linked with lower completion of research visits^35,36. Other limitations include the incomplete response rate in some of the questions including 13.3% of patients choosing not to answer the PRAPARE item that asked if there were any needs. Factors contributing to incomplete responses included reluctance to disclose financial information and apprehension regarding the potential impact of survey responses on custody cases, even though families were assured of the confidentiality of their responses.

In conclusion, our study suggests that there is a relationship between parental HL and disease activity in patients with JIA. Given that less patient portal utilization and higher no-show rate are associated with limited HL, these may represent areas of interventions for improving outcomes in patients with JIA. Since families with limited HL have less social support, they may benefit more from support groups and encouraging communities and networks. Next steps include implementation of standard screening of HL in all JIA patients. When identifying parents with limited HL, a potential intervention would include giving a video that summarizes important aspects of healthcare interaction such as how to activate and trouble shoot patient portals and how to complete a patient assistance program application for JIA related therapies.