Prospective quality of life outcomes in pediatric fecal incontinence following bowel management

Abstract

Background

Severe fecal incontinence (FI) is common in patients both with and without anorectal malformations. Whether a formal bowel management program (BMP) has significant effects on FI, psychosocial development of the child, and caregiver stress is poorly understood. We hypothesize that BMP participation results in long-term clinical and quality of life (QOL) improvements for patients and caregivers.

Methods

Using a prospective cohort study over three years, 342 children (age 3–12 years) and caregivers were followed for one year after attending a week-long BMP, during which a regimen was tailored to promote daily stool evacuation.FI QOL was measured with the validated Cincinnati Fecal Incontinence Scale (CINCY-FIS), evaluating multiple subscales, including parental stress. Scores were obtained at multiple timepoints following BMP (baseline, 2 weeks, 3 months, 1 year).

Results

Within 2 weeks, BMP participation significantly improved FI with increased frequency of daily daytime voluntary bowel movements (20%–70%, p < 0.001) and decreased daily daytime and nighttimeinvoluntary bowel movements (60%–20%, p < 0.001; 30%–10%, p < 0.05). Marked improvements in CINCY-FIS were observed across multiple QOL subscales, with the greatest in parental stress, and sustained through one year.

Conclusions

BMP results in significant and sustained improvement in FI and QOL for patients and caregivers.

Introduction

Pediatric fecal incontinence (FI), with an estimated prevalence of 4.4% [1], is distinct from adult FI and is variable in etiology and severity. In the pediatric population, FI may be due to true FI, in which there is loss of normal continence mechanisms (frequently from congenital anorectal malformations), or overflow pseudo-incontinence in which involuntary stool leaks due to fecal impaction. Additionally, the effects of pediatric FI extend to their caregivers and impact the entire family. Previous studies have shown that parents of children with FI report significantly lower quality of life (QOL) scores compared to their peers [2, 3]. Specifically, parents of children with FI have been found to experience stress at the most upper-bound limits of the general population [2]. However, previous studies used generic QOL measures (i.e. PedsQL4.0) that do not capture disease-specific challenges, nor assess parental stress [4].

To address this gap, our group previously designed and validated the Cincinnati Fecal Incontinence Scale (CINCY-FIS) [5]. A brief caregiver report of FI-specific QOL in children, the CINCY-FIS is a psychometrically validated tool that quantifies the physical and psychosocial impact of inappropriate passage of stool (whether true FI or overflow pseudo-incontinence) on both the patient and caregiver (Appendix 1). The Pediatric Fecal Incontinence Severity Score (PFISS) is our internally created measure developed to quantify various aspects of pediatric FI, such as frequency of daytime and nighttime involuntary bowel movements (IVBM). To date, no studies have utilized these or similar tools to assess the long-term effects of pediatric FI interventions.

Patients with severe pediatric FI refractory to routine management are typically referred to intensive bowel management programs (BMP). Participation in a BMP requires a considerable amount of time and energy. Patients are evaluated by a multidisciplinary team prior to beginning a laxative or enema-based regimen. The choice of regimen is multifactorial and depends not only on the underlying diagnosis and severity of FI, but also patient and parental goals. Daily assessments are then performed throughout the course of one week often away from work and home, with the goal of emptying the rectum and distal colon at least once a day. After completing one week of bowel management, patients are either seen in follow-up appointments as needed, or are followed remotely to allow for regimen adjustments independent of a clinic visit. When parents commit to this significant endeavor, however, there are no studies demonstrating long-term outcomes with disease-specific measures to help predict and manage expectations of BMP participation for both the patient and their caregivers.

The short term and intermediate outcomes of BMP have been described [4, [6], [7], [8]] yet there are no long-term prospective studies using scales specific to children [2, 4]. Thus, measures of clinical success vary widely in the literature [9]. For example, a 24-h period without involuntary bowel movements (IVBM) during a week of BMP was considered successful, while others used improvement in the adult Wexner score to identify success [4]. This variation underscores the need for studies utilizing disease-specific QOL tools to critically evaluate BMP effect on pediatric FI. Our study aims to address the gap by administering the CINCY-FIS, combined with PFISS, in a prospective manner to assess the clinical and QOL outcomes at 1 year after completion of a BMP in the pediatric population.