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Original Articles: Gastroenterology

Lack of Knowledge and Low Readiness for Health Care Transition in Eosinophilic Esophagitis and Eosinophilic Gastroenteritis

Eluri, Swathi; Book, Wendy M.; Kodroff, Ellyn; Strobel, Mary Jo; Gebhart, Jessica H.; Jones, Patricia D.§; Menard-Katcher, Paul||; Ferris, Maria E.; Dellon, Evan S.

Author Information
Journal of Pediatric Gastroenterology and Nutrition: July 2017 - Volume 65 - Issue 1 - p 53-57
doi: 10.1097/MPG.0000000000001415

Abstract

Erratum

Two articles published in Journal of Pediatric Gastroenterology & Nutrition in 2017 did not include relevant patient advocacy groups in the funding information in the article footnotes.

In the article that appeared on page 546 of the April 2017 issue, “Propofol Use in Pediatric Patients With Food Allergy and Eosinophilic Esophagitis,” as well as the article that appeared on page 53 in the July 2017 issue, “Lack of Knowledge and Low Readiness for Health Care Transition in Eosinophilic Esophagitis and Eosinophilic Gastroenteritis,” the final sentence of the funding information should have read:

“This study was also funded by U54 AI117804, which is part of the Rare Disease Clinical Research Network (RDCRN), an initiative of the Office of Rare Disease Research (ORDR), NCATS, and is funded through collaboration between NCATS, NIAID, and NIDDK, as well as the patient advocacy groups American Partnership for Eosinophilic Disorders (APFED), CURED, and the Eosinophilic Family Coalition (EFC), which have collectively resulted in the Consortium of Eosinophilic Gastrointestinal Disease Researchers.”

These articles have been corrected online.

Journal of Pediatric Gastroenterology and Nutrition. 66(4):709, April 2018.

What Is Known

  • Eosinophilic esophagitis and eosinophilic gastroenteritis are chronic immune-mediated conditions that affect both children and adolescents.
  • Health care transition needs in eosinophilic esophagitis/eosinophilic gastroenteritis have not been well described or formally assessed.

What Is New

  • This is the only study to assess health care transition knowledge and readiness in eosinophilic esophagitis/eosinophilic gastroenteritis.
  • Knowledge of health care transition was lacking in >75% of patients with eosinophilic esophagitis/eosinophilic gastroenteritis and in parents of patients with these conditions.
  • Health care transition readiness scores measured with the Self-Management and Transition to Adulthood with Rx Questionnaire in patients aged 13 to 25 years and parents were notably low compared with other chronic illnesses.

Eosinophilic esophagitis (EoE) and eosinophilic gastroenteritis (EGE) are chronic, immune-mediated conditions (1–5) affecting both children and adolescents/young adults (6–9). Although the natural history of these diseases is not well understood, there is evidence (10) that children with EoE continue to be symptomatic as they transition to adulthood and require ongoing therapy and chronic disease management. Our clinical experience is that the same is true of EGE.

Health care transition (HCT) is the “purposeful, planned movement of adolescents and young adults with chronic physical and medical conditions from child-centered to adult-oriented health care systems” (11,12). The key to successful HCT is to accurately measure HCT readiness to determine the most optimal time and mode of transitioning to an adult provider (13). HCT needs in adolescents with chronic conditions has been recognized and studied to facilitate the development of needs-based HCT programs (14–18). There are, however, certain health conditions such as EoE/EGE in which HCT needs have not been well described (19) or formally assessed in a population with high disease burden.

Therefore, we aimed to assess whether patients with EoE/EGE and parents of patients with these conditions had knowledge of HCT, and to determine the scope and predictors of HCT knowledge. The secondary aim was to measure HCT readiness in adolescents/young adults with EoE/EGE using the Self-Management and Transition to Adulthood with Rx (STARx) Questionnaire (13,20) and to correlate it with factors impacting transition readiness.

METHODS

Study Design and Population

This was a cross-sectional survey of patients who were 13 years or older with EoE/EGE or parents of patients with EoE/EGE of all ages. Of note, these were not parent-child pairs. Using the Qualtrics web-based platform, we deployed an online survey through the American Partnership for Eosinophilic Disorders (APFED) and Campaign Urging Research for Eosinophilic Disease (CURED) Web sites, email lists, social media sites, and other online resources. Two versions of the survey were administered: patient-reported and parent-reported. Patients answered the questions themselves. Parents were requested to answer questions regarding their children and their own familiarity with HCT. The study was approved by the University of North Carolina Institutional Review Board before study initiation and informed consent/assent was obtained from all study participants.

STARx Health Care Transition Readiness Assessment

All participants completed a STARx Questionnaire (13) to assess readiness to transition from pediatric to adult health care services. Patients completed the survey by answering questions about their own HCT readiness and parents regarding perceived HCT readiness of their children. The STARx Questionnaire measures self-reported HCT readiness and self-management for adolescents/young adults with chronic health conditions. The development, reliability, and validity of the tool have been described previously (13,20). In brief, the STARx Questionnaire comprises 18 questions measured on a 5-point Likert scale and measures HCT readiness in 6 domains, including medication management, provider communication, engagement during appointments, disease knowledge, adult health responsibilities, and resource utilization. The questionnaire is scored from 0 to 90, with higher scores indicating higher levels of HCT readiness.

Statistical Analysis

Descriptive statistics were used to summarize characteristics of the study population. Bivariate analyses were used to compare those who did and did not have prior knowledge of HCT in the subset of patients diagnosed with EoE/EGE when 25 years or younger. Student t test and Wilcoxon rank sum were used for continuous variables and Pearson χ2 test was used for categorical variables. HCT readiness analysis using the STARx Questionnaire was restricted to adolescents/young adults aged 13 to 25 years as it was validated in this age group (13,20). Analysis of the parent-reported survey was similarly restricted to those who had children with EoE/EGE aged 13 to 25 years. Mean HCT readiness in each of the 6 domains and mean total HCT readiness scores were calculated from both the patient and parent surveys. Differences between the parents’ perceptions of their children's readiness and patient self-reported readiness were compared using Student t test. Association between HCT readiness scores and patient and provider characteristics were calculated and differences in scores within each domain with increasing age groups were also calculated using analysis of variance. Differences were considered statistically significant at an alpha level <0.05. All analyses were performed using STATA 13 (StataCorp, College Station, TX).

RESULTS

A total of 450 participants completed the survey: 205 patients and 245 parents. We restricted the analysis to those diagnosed with EoE/EGE at age 25 years or younger, resulting in a sample of 75 patients and all 245 children of parent respondents. A majority in both groups had EoE (Table 1). Among those who took the patient survey, 20% were male compared with the parent survey in which 70% of the children with EoE/EGE were boys. In the patient survey, median age (23 ± 7 vs 12 ± 6) and age of disease onset (16 ± 6 vs 8 ± 5) was higher, and a larger proportion had active symptoms (92% vs 77%) compared to the children of parents who took the parent survey. Approximately 80% of the subjects with EoE/EGE in both groups were on active dietary therapy for disease management. A very high proportion from both groups was under the active care of a primary care provider (PCP) (84% vs 97%), a gastroenterologist (GI) (85% vs 92%), a nutritionist (69% vs 71%), and an allergist (84% vs 89%).

Table 1
Table 1:
Participant characteristics

Overall, 78% (n = 52) of the patients and 76% (n = 187) of parents reported having no prior knowledge of HCT. Factors associated with lack of HCT knowledge among patients were older age (24 vs 19 years, P = 0.03), older age at disease diagnosis (17 vs 12 years, P < 0.01), and already seeing an adult gastroenterologist (63% vs 38%, P < 0.05) (Table 2). Few factors were associated with parental unfamiliarity with HCT other than active steroid use. For example, parents who had children with EoE/EGE and who were on active steroid treatment were less likely to know about HCT (48% vs 31%, P = 0.03). Knowledge regarding HCT was not associated with a difference in preferred age to discuss or initiate the transition process. Interestingly, the mean preferred age to initiate HCT was at least 18 in all groups and the highest preferred age of 24 years was in participants taking the patient survey who were familiar with HCT (Table 2).

Table 2
Table 2:
Association of patient and provider characteristics and familiarity with HCT in patients and parents of patients with eosinophilic esophagitis and eosinophilic gastroenteritis

The proportion of participants who reported knowledge of HCT between the ages of 0 to 25 years were divided into 5 age groups (Supplemental Digital Content 1, Figure, https://links.lww.com/MPG/A786). Among those who took the patient survey, only about half of those aged between 16 and 20 years were familiar with HCT. The largest percent of parents who were familiar with HCT had children aged 11 to 15, but this was still under 50%. HCT knowledge was similarly low among parents with children aged 0 to 10 and 15 to 25 years.

Health Care Transition Readiness

Out of 50 subjects aged 13 to 25 and 123 parents of patients in this age range, mean HCT readiness score was 30.4 ± 11.3 with scores in domains of provider communication, disease knowledge, and engagement during appointments being somewhat higher than other domains (Table 3). Mean parent-reported HCT readiness score was 35.6 ± 9.7 with higher scores in the domains of medication management and disease knowledge and lower scores in the adult health responsibilities and resource utilization domains. Overall, there was a significant difference in parent-reported HCT readiness scores and patient-reported scores across all domains except for disease knowledge and adult health responsibilities. Parent-reported readiness of their children in the domains of provider communication (8.2 vs 5.8, P < 0.01), engagement during appointments (9.7 vs 5.4, P < 0.01), and resource utilization (6.3 vs 4.4, P < 0.01) was significantly lower than patient-reported readiness. For medication management, patient-reported readiness scores were lower than the parent-reported scores for their children (6.5 vs 8.8, P < 0.01).

Table 3
Table 3:
Health care transition readiness scores using the STARx Questionnaire in adolescents/young adults with eosinophilic esophagitis and eosinophilic gastroenteritis

There was no association between overall HCT readiness score and disease type, age, age at diagnosis, percent of life with disease, active symptoms, active steroid use, dietary therapy, or provider type (Supplemental Digital Content 2, Table 1, https://links.lww.com/MPG/A787). When adolescents/young adults were divided into age groups (13–15, 16–18, 19–21, and 22–25 years of age) based on developmental milestones, there were no significant differences between HCT readiness scores among the different age groups within each domain other than adult health responsibilities and engagement during appointments (Supplemental Digital Content 2, Table 2, https://links.lww.com/MPG/A787). Interestingly, participants taking the patient survey who were 22 to 25 had lower readiness scores than those in the 13 to 15 age group in the domain of adult health responsibilities (2.8 vs 4.7, P = 0.01). In the parent survey, there was a statistically significant increase in perceived transition readiness of their children with increasing age in the domain of engagement during appointments (7.5 vs 5.0, P = 0.01).

DISCUSSION

Our study is the only of its kind to assess HCT knowledge and readiness in EoE/EGE. Knowledge of health care transition was lacking in >75% of the participants, and this lack of HCT knowledge was similar in patients with EoE/EGE and in parents of patients with these conditions. This is a significant deficit in knowledge, particularly as this survey was conducted among a group of subjects connected to patient advocacy groups and support web sites, who were likely highly motivated. Among parents, age of the children did not correlate with transition knowledge. In fact, a similar proportion of parents who had children aged 0 to 10 years reported being familiar with HCT as those who had children aged 15 to 25. In addition, HCT readiness scores measured with the validated STARx Questionnaire were notably low in adolescents/young adults with EoE/EGE. Overall, there was no correlation between overall HCT readiness scores and disease type, markers of disease severity, age, disease duration, or provider type.

When comparing EoE/EGE to other chronic diseases, we were not able to make a valid comparison of the lack of HCT knowledge because HCT knowledge can be assessed in multiple ways. The preferred age for HCT of 18 years in both patients and parents reported in our sample is, however, similar to those of other non-GI chronic diseases (21) and inflammatory bowel disease (IBD) (22,23). Even though pediatric societies recommend ages closer to 12 for HCT initiation (24), the age of 18 is likely seen by patients and parents as a more practical choice as it coincides with a social transition to secondary schooling such as college (25). Another similarity was that the higher readiness scores in domains such as “provider communication” and “engagement during appointments” seen in our EoE/EGE population were also areas of higher mastery in the IBD population (26). One major difference was that the mean scores for HCT readiness were considerably lower in EoE/EGE compared with other chronic disease conditions (13). In a sample comprised primarily of adolescents/young adults with other chronic conditions (chronic kidney disease, IBD, cystic fibrosis, and systemic lupus erythematous) mean HCT readiness scores measured by the STARx Questionnaire ranged from 43 to 59 with an increase in readiness scores with increasing age of the participants (13).

One of the reasons for lower HCT readiness in EoE/EGE compared with other chronic conditions could be due to lack of HCT knowledge. Three quarters of a highly motivated group of patients with EOE/EGE and parents in the present study lacked HCT knowledge, and this is markedly higher than has been reported in other conditions (27). There are also provider-reported barriers to HCT that have been identified (23) because the process of HCT is a collaborative effort between patients, caretakers, and health care providers. Provider-perceived barriers to HCT such as lack of infrastructure, disease knowledge, and training in adolescent care (23,28) have not been assessed in EoE/EGE and would be a valuable perspective to ascertain in future studies.

HCT needs to be prioritized because of the increasing incidence and prevalence of EoE (7). Studies have shown that the prevalence of EoE decreases after age 45 and a majority affected is children and adolescents/young adults (9,29,30). For EGE, the overall prevalence is lower compared with EoE, but EGE is most commonly diagnosed in children younger than 5 years (4). As a result, there will likely be a large influx of patients with EoE/EGE who will need to transition to adult providers without disruption of health care. Because of a dearth of literature on this topic in EoE/EGE (19), HCT programs that are tailored to these diseases must be developed and studied. Unlike other chronic diseases, there are certain aspects of health care that are specific to patients with EoE/EGE (19). This includes HCT not just from a single pediatric to an adult provider but transitioning to an adult multidisciplinary team including nutritionists and allergists, because patients with EoE/EGE often require a collaborative team for optimal management. In addition to medical and dietary treatment, there is also a need for serial endoscopic assessments with potential need for interventions such as dilations and biopsies that can add to the complexity of HCT.

Our study has multiple strengths and some weaknesses. First, it is the only study to have evaluated HCT knowledge and readiness in a sample of patients with EoE and/or EGE. Second, HCT readiness was measured with the disease-neutral, self-reported STARx Questionnaire, which has been shown to be a valid self-report tool with high reliability that can be used to assess transition readiness and self-management skills in adolescents/young adults with a variety of health conditions (20). Third, the study was conducted using an online survey that increases the likelihood of providing a diverse sample of patients with EoE/EGE, though there could be selection bias. Those who participated in the study were most likely a highly motivated group of patients and caregivers who have access to an online interface and possibly more social support. In this highly selective group of activated health consumers, we would, however, expect a higher proportion with HCT knowledge and larger readiness scores, which we did not find in our study. Therefore, it is possible that our results actually overestimate HCT knowledge in the general EoE/EGE population, highlighting an area that needs significant attention. We also did not obtain information regarding factors that affect HCT such as health insurance, race, access to resources, and education level. Because the survey was distributed through multiple online resources, we were unable to determine the overall survey response rate. In addition, provider perspectives were not collected. Finally, as the study was cross-sectional, we do not have longitudinal information regarding change in HCT readiness over time or outcomes that correlate with HCT knowledge and readiness.

In summary, approximately three-quarters of patients and parents of children with EoE/EGE who were diagnosed with the disease at 25 years of age or younger were unfamiliar with health care transition. Given our methodology, this rate of unfamiliarity may be an underestimate. In addition to the significant lack of knowledge of HCT, HCT readiness was also low in adolescents/young adults compared with other chronic diseases at baseline without any interventions. Given that a large cohort of patients with EoE/EGE will need to transition to adult providers, HCT preparation and readiness assessments should become a priority in these patients and their parents. In addition, barriers for knowledge need to be identified to improve the process of transitioning from pediatric to adult care in the EoE/EGE population.

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Keywords:

eosinophilic esophagitis; eosinophilic gastroenteritis; health care transition readiness

Supplemental Digital Content

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