What Is Known
- Diet treatment is effective in resolving symptoms and inflammation related to eosinophilic esophagitis.
- Adherence to diet can be difficult in some patients with eosinophilic esophagitis.
What Is New
- Issues related to dietary treatment of children with eosinophilic esophagitis creates caregiver and child stress.
- Stressful factors relate, not only to food preparation and cost, but also concerns regarding mealtime structure and exposure to food allergens.
Eosinophilic esophagitis (EoE) has become one of the most common causes of feeding problems in children (1). Management of EoE can involve dietary restrictions, and in some cases, placement of a gastrostomy tube (G-tube). Consequently, children and families can experience decreased health-related quality of life (HRQoL) that often negatively affects the typically pleasurable activity of eating (2).
EoE-related stress can also be associated with a delay in diagnosis, persistent symptoms, unsuccessful trials of medications, treatment adverse effects, dietary restrictions, financial hardships associated with specialized diets and formulas, and the social stigma of eating differently than others (3). One recent study examined 163 caregivers (CGs) of children with all eosinophilic gastrointestinal diseases and found that CG stress was associated with psychological distress, income, child behavioral problems, treatments, and disease severity (4). To date, stress experienced by CGs of patients with only EoE has not been examined.
To begin to address this, we administered a series of surveys to CGs to measure anxiety and stress that was encountered as a part of daily life. The aim of the present study was to identify the most stressful elements experienced by CGs of children with EoE.
During the American Partnership for Eosinophilic Diseases (APFED.org) annual patient education conference, CGs of children between the ages of 2 and 18 years and youth with a diagnosis of EoE were recruited and compensated to complete paper-and-pencil questionnaires.
CGs responded to 3 questionnaires that asked directly about their emotional state/degree of stress and 3 questionnaires in which CGs served as parent proxy by reporting on their child's emotional state. Youth were administered 3 questionnaires.
CG questionnaires included State-Trait Anxiety Inventory (STAI), STAI-Form Y (5). STAI assesses both state anxiety (S-Anxiety) by asking how respondents feel “right now” and trait anxiety (T-Anxiety), which evaluates stable aspects of “anxiety proneness”; Food Allergy Quality of Life-Parental Burden (FAQoL-PB) questionnaire (6). FAQoL-PB is a 17-item measure that uses a 7-point Likert scale ranging from 1 (not troubled) to 7 (extremely troubled). Questions assess burden of food allergies as they relate to CG's perceptions of meal preparation, social activities, and food allergy–related worries and anxieties during the previous week; EoE Caregiver Stress Questionnaire (EoE-CGSQ). EoE-CGSQ was developed by the authors, a gastroenterologist (G.T.F.), allergist (D.A.), pediatric psychologist (J.R.), and a social worker (C.C.). EoE-CGSQ is a nonvalidated, 23-item questionnaire that was developed to capture child health information (eg, length of time to diagnosis) and areas of stress as reported by CGs caring for children with EoE. It includes 15, 5-point Likert-scale items (1 = not at all to 5 = severe) and 8 dichotomous (yes/no) items (see Supplemental Digital Content, Questionnaire, http://links.lww.com/MPG/A861).
Youth and CGs as proxies responded to 3 questionnaires: Pediatric Quality of Life Eosinophilic Esophagitis Module Version 3.0 (PedsQL EoE, 3.0) (7) includes 33-items developed for children ages 2 to 18 years and assesses EoE-specific HRQoL. Higher scores indicate better HRQoL; Revised Child Anxiety and Depression Scale (RCADS) (8) (RCADS is a 47-item, self-report instrument and was selected because it measures specific elements of anxiety and depression that potentially correlate with disease-specific areas of functioning); and Screen for Child Anxiety Related Disorders (SCARED) (9). SCARED asks directly about specific features of anxiety and uses a 3-point Likert scale (0 “not true or hardly ever true” to 2 “very true or often true”).
Raw scores were entered into SAS 9.4 for analysis and when appropriate converted to standardized scores according to the measure used. Pearson correlations were calculated to assess the relation between CG and youth psychological distress, EoE-HRQoL, and EoE-specific stress factors.
Forty-six families were recruited and 38 CGs, primarily Caucasian (92%) and mothers (84%) ranging in age from 34 to 44 (39.4: ±4.9 years, SD) years completed surveys (Table 1). Seventeen youth (mean age 11 years ±2.5 SD) participated. The majority of children (N = 38, 63% boys) experienced a diagnostic delay of more than 12 months before receiving a diagnosis of EoE and 41% waited 24 months or longer. Results from the PedsQL-EoE assessment, in which higher scores indicate better HRQoL, demonstrated that patients with EoE identified Food/Eating; Food/Feelings; and Treatment as 3 areas most affecting HRQoL. Similarly, parent proxy reports also identified Food/Eating and Food/Feelings as most negatively affecting children's HRQoL (Table 2). When examining stressful elements in patients’ and CGs’ lives, issues related to mealtimes and food were prominent. For instance, moderate to severe CG stress was associated with buying and preparing separate foods/meals to fit children's dietary requirements, cost of foods to fit dietary requirements, and disruption of family structure at mealtimes (Table 3). To determine the relation of mealtimes and food-related issues with anxiety, worries, and HRQoL, we performed a series of correlations.
To assess the relation between CG anxiety and food-related factors, correlations between CG STAI-Form Y (S-Anxiety) and the EoE-CGSQ questions were conducted. We found that cost of food [r(38) = 0.54, P = 0.0005], preparation of food [r(38) = 0.38, P = 0.02], and family mealtime structure [r(38) = 0.42, P = 0.01] all correlated with CG State anxiety. Parental burden of meal preparation, social activities, and food allergy–related worries as determined by the FAQoL-PB total scores correlated with CG STAI Form Y (S-Anxiety) scores [r(38) = 0.41, P = 0.03]. Results from the FAQoL-PB questionnaire also determined that CG food-related burden (mean question score = 3.2) was similar to scores typically attributed to the stress of managing a child with 3 or more anaphylactic food allergies (6). Diminished HRQoL among youth correlated with increased anxiety and depression [PedsQL-EoE score correlated with both SCARED total score, r(17), −0.69, P = <0.004 and RCADS r(17), −0.75, P = <0.0005]. In fact, 50% of youth reported high-frequency worry, anger, and sadness related to specialized diets as measured by the PedsQL-EoE subscale of Feelings (Mean x = 47).
In the present study of psychosocial stressors associated with childhood EoE, we found that factors leading to CG anxiety and stress were related to cost of specialized diets, meal preparation, and mealtimes. Our results suggest that providers should consider the effect that dietary treatment of EoE may bear upon the family and the unidentified burden associated with this intervention. The present study did not inquire about a pre-existing diagnosis of CG/youth anxiety or depression; however, our results would suggest that regardless, additional psychosocial support may be needed if diet restrictions are recommended.
Our findings add to a growing body of literature focusing on comorbid issues related to EoE. With increasing experience, more knowledge is developing regarding not only the natural history of EoE, but also quality of life with the disease and adverse effects associated with treatment. Although previous studies of EoE and youth HRQoL have assessed the effect of restricted diets in the treatment of EoE (7), results of our study underscore practical, financial, and psychological burdens associated with specialized diets and the effect on CGs. Here we discovered that CG stress was linked to cost of food and preparation, lack of structure at mealtimes, social aspects of eating, and coping with large numbers of allergens. Finally half of youth were affected by worry, anger, and sadness related to specialized diets. Each of these areas of concern can readily be addressed with CGs and patients during decision making for EoE treatments.
Our study was limited by the fact that this was a patient/CG-based survey and less than half of CGs had corresponding youth reports. Therefore, comparison of patient experience compared with CG experience proves to be tenuous. Moreover, clinical confirmation of a diagnosis was not possible, and specific diet restrictions were not assessed. It is likely that respondents were highly motivated to participate because they were already attending an out-of-town patient education conference. Therefore, results may not reflect the entire EoE-related population at large but do provide critical insights into the lives of families who are faced with adhering to diets that could successfully affect clinical and histological features of their disease. Moreover, the majority of patients in our study had been under treatment (eg, a known diagnosis for >12 months) and therefore their symptom severity could have been attenuated at the time of the study due to established interventions. Future studies should assess children and families within the first 6 months of diagnosis while evaluating symptom severity and perceived stressors, something the present study did not allow for as we did not inquire about symptom severity. Established care and intervention could also explain why youth did not list symptoms as 1 of the top 3 factors affecting HRQoL. Finally, we used a nonvalidated tool to assess EoE-specific stress. Because EoE is a relatively new disease, we felt an assessment tool to address features related to EoE was needed to begin to address this emerging issue. Future efforts and studies will be needed to develop validated tools to assess stress more completely in patients and CGs with EoE.
To enhance adherence and improve outcomes, providers should carefully consider and discuss youth and CG attitudes, resources, and motivation when prescribing EoE-related diets. Such discussion will lead to improved clinical care and enhanced provider/patient communication regarding interventions.
The authors thank the patient and parent participants and the American Partnership for Eosinophilic Disorders for their cooperation with the present study.
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