Diet and bowel function in children with Hirschsprung’s disease: development and content validation of a patient-reported questionnaire

Abstract

Background Although dietary adjustments are recommended frequently for bowel symptoms, evidence of diet’s impact on bowel function is lacking. The aim was to develop a patient-reported outcome instrument, for children with and without Hirschsprung’s disease (HD), to explore experiences of dietary effects on bowel function. Methods Children with and without HD and their parents participated. Questionnaire items regarding the impact of diet on bowel function originated from focus group discussions. Specific food items, reported in the literature or in focus groups to cause bowel functional effects, were listed requesting each item’s effect size and effect type. Content validity was tested within two separate semistructured interviews. A pilot test was performed. Assessing comprehension, relevance and wording clarity structurally, revisions were made accordingly. Children’s bowel function was assessed through the validated Rintala Bowel Function Score. Results A total of 13 children with and without HD, median age 7 (range 2–15) years, and 18 parents participated in the validation. Each question’s relevance had been ranked highly early in the validation process but most questions needed refining for improving clarity and comprehension. Wordings regarding bowel symptoms and emotions connected to food in particular were perceived to be sensitive and complex. Specifically wording regarding some bowel symptoms (gases, pain) and parental stress emotions (guilt, ambivalence) were, consistent with participants’ opinions, subjected to multiple step revisions. Following the validation process, which included two semistructure interviews with different participants and then a pilot test with a third cohort, a full track overview of changes and rewording made in all steps of the validation process was presented. The final questionnaire then comprised 13 questions assessing foods’ significance for bowel function, emotions, social impact and 90 specific food items’ possible effects and effect sizes on bowel function. Conclusions The Diet and Bowel Function questionnaire, enabling answering by children, was developed and the content validated qualitatively. This report presents insights into the whole validation process, declaring reasons for the selected question- and answering options, and their wordings. The Diet and Bowel Function questionnaire can be used as a survey questionnaire to enhance understanding of dietary effects on bowel function in children, and its results can be supportive in improving dietary-treatment programs.

A Narrative Review of Patient-Reported Outcome Measures and Their Application in Recent Pediatric Surgical Research: Advancing Knowledge and Offering New Perspectives to the Field

“…Out of 345 articles identified by the database searches, 49 articles met the inclusion criteria. 28 29 30 31 32 33 34 35 36 37 38 39 40 41 42 43 44 45 46 47 48 49 50 51 52 53 54 55 56 57 58 59 60 61 62 63 64 65 66 67 68 69 70 71 72 73 74 75 76 Most concerned esophageal atresia ( n = 17) and Hirschsprung's disease ( n = 14). None of the articles focused on duodenal atresia or omphalocele.…”

“…The 49 articles identified a total of 29 PROMs. 66 77 78 79 80 81 82 83 84 85 86 87 88 89 90 91 92 93 94 95 96 97 98 99 100 101 102 103 104 105 The most frequently used PROM, applied predominantly in patients with esophageal atresia ( n = 11) or Hirschsprung's disease ( n = 8), was the Pediatric Quality-of-Life Inventory 4.0 generic core scales (PedsQL 4.0) 83 ( n = 31). Four common assessments for colorectal function, including (in)continence, in anorectal malformations and/or Hirschsprung's disease were the Rintala Bowel Function Scoring System 77 ( n = 9), Baylor Continence Scale (BCS) 78 ( n = 4), Cleveland Clinic Fecal Incontinence Severity Scoring System (or Wexner score) 81 82 ( n = 2), and the Krickenbeck criteria 102 ( n = 1).…”

“…One study was dedicated to the development of a specific PROM, known as the Diet and Bowel Questionnaire. 66 Four studies focused on the psychometric evaluation of one condition-specific PROM (Esophageal-Atresia-Quality-of-life Questionnaires, EA-QOL) across four countries. 37 47 49 50 None of the PROMs were subjected to an evaluation of their impact on patient, process, and/or health service-related outcomes.…”

Self-Reported Effects of Diet on Gastrointestinal Symptoms in Healthy Children

“…Gastrointestinal symptoms were assessed by questions about constipation (Rintala Bowel (18). This instrument was developed for children aged 1-18 years old and their parents.…”

Children with Hirschsprung’s Disease Report Dietary Effects on Gastrointestinal Complaints More Frequently than Controls

“…In our focus group study, diet was shown to play a key role in parental self-treatment of GI symptoms in children with HD, and such changes have an impact on daily life [ 12 ]. To increase knowledge on diet’s role in bowel function, we developed the Diet and Bowel Function Questionnaire [ 13 ]. In healthy children who answered the questionnaire, any GI effects as a result of diet were only evident among 52%, and the reported effects were weak [ 14 ].…”