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Stressful Life Events in Children With Functional Defecation Disorders

Philips, Elise M.*; Peeters, Babette*; Teeuw, Arianne H.; Leenders, Arnold G.E.; Boluyt, Nicole§; Brilleslijper-Kater, Sonja N.; Benninga, Marc A.*

Author Information
Journal of Pediatric Gastroenterology and Nutrition: October 2015 - Volume 61 - Issue 4 - p 384-392
doi: 10.1097/MPG.0000000000000882

Abstract

Constipation is a common problem in children with worldwide prevalence rates ranging from 0.7% to 29.6% and may have a great influence on a child's health and behavior (1). These children usually present with infrequent, large, and painful defecation often accompanied by the involuntary loss of feces in their underwear. In the majority of children, the loss of feces is the result of severe rectal fecal impaction. In approximately 20% of the children, fecal incontinence (FI) is a sole symptom without any other symptom of constipation and classified as functional nonretentive fecal incontinence (FNRFI). Both conditions are considered to be functional defecation disorders once organic causes are ruled out (2). Long-term follow-up studies in children with severe constipation and FNRFI showed that despite intensive medical and/or behavioral treatment symptoms persist into adulthood in 15% to 30% of children (3,4).

The pathophysiology of functional defecation disorders is probably multifactorial. Factors such as genetic predisposition, low socioeconomic status, and most frequently the development of withholding behavior after experiencing a painful or frightening evacuation have been proposed as factors leading to functional defecation disorders (5,6).

Several studies in adults have shown that the experience of stressful life events, including child (sexual) abuse during infancy and childhood, is an important risk factor for the development of functional bowel disorders in adulthood (7). Stressful life events including child (sexual) abuse are unfortunately common in childhood and could therefore be a major risk factor for developing functional bowel disorders in children.

More knowledge about the prevalence of stressful life events including child (sexual) abuse in children with functional defecation disorders could contribute to a better insight into the pathophysiology of these disorders that are often hard to treat.

In this review, we aim to investigate and summarize the prevalence of stressful life events including (sexual) abuse in children with functional defecation disorders by conducting a systematic review of the literature.

METHODS

Search Strategy

A literature search was performed to identify studies investigating the prevalence of child (sexual) abuse and the experience of stressful life events in children presenting with functional defecation disorders. MEDLINE, EMBASE, and PsycINFO were searched from inception to August 2014 using keywords (medical subject headings [MeSH]terms and text words) as described in Supplemental Table 1 (Supplemental Digital Content 1, https://links.lww.com/MPG/A509). In addition, reference lists of reviewed articles and included studies were hand searched for other relevant articles. Studies in any other language than English, Dutch, or German were excluded from the analysis.

Study Selection

Three reviewers (E.M.P., B.P., S.N.B.-K.) independently screened the titles and abstracts of all articles retrieved by the above-mentioned search for eligibility. All relevant studies, as well as studies for which the abstracts did not provide sufficient information for inclusion or exclusion, were retrieved as full papers. To assess eligibility, the following 3 inclusion criteria were applied: the study was a cohort, case-control, or cross-sectional study; the study population consisted of children 3 to 18 years of age with functional defecation disorders (ie, functional constipation with or without FI or FNRFI), compared with a matching control group without functional defecation disorders or just a population of children 3 to 18 years of age with functional defecation disorders, but without a matching control group; and the prevalence of child abuse or traumatic and/or stressful life events in children with functional defecation disorders had to be one of the aims and outcome measures. Studies of patients with defecation disorders as a result of organic causes or medication were excluded.

Quality Assessment

To assess methodological quality and risk of bias of the included studies we used the guidelines and checklist for appraising a medical article from Fowkes and Fulton (8). To evaluate the reporting quality of the included studies, we developed a quality list based on The Strengthening the Reporting of Observational Studies in Epidemiology (STROBE) statement (9,10). Two reviewers (E.M.P., B.P.) independently rated these 2 checklists. The guidelines and checklist for appraising a medical article according to Fowkes and Fulton is depicted in Table 1(11–18). Each criterion has been assigned as major (++), minor (+), or no problem (0) in terms of their expected effect on the results. The reporting quality has been assessed with the 11 items of the reporting quality score list (Table 2) (11–18). Each of the items had 3 answer options: “yes”/“no”/“do not know.” No points were given to the items because this is not a standardized quality list. Disagreements were resolved through consensus.

T1-4
TABLE 1:
Guidelines and checklist for appraising a medical article according to Fowkes and Fulton (8)
T2-4
TABLE 2:
Reporting quality of the included studies

Data Extraction

Two reviewers (E.M.P., B.P.) independently performed an individual data extraction from the original reports of all included studies. Extracted data included study design, participants, setting, definition of constipation, FI or FNRFI, outcome measures, and results. These data were reported in a descriptive way.

RESULTS

The search generated 946 titles and abstracts (Supplemental Figure 1, Supplemental Digital Content 2, https://links.lww.com/MPG/A510) of which 36 studies were judged relevant and retrieved for full text. As depicted, 28 of 36 articles were excluded for various reasons (19–46). Of the 8 studies that were included, 7 studies focused on the prevalence of stressful life events (11–17), whereas only 1 study measured the prevalence of child (sexual) abuse (18). All included studies were cross-sectional studies (11–18). Full characteristics of these studies are described in Table 3(11–18). Complete data are described in Supplemental Table 2 (Supplemental Digital Content 3, https://links.lww.com/MPG/A511).

T3-4
TABLE 3:
Study characteristics and results (only the significant data included)
T4-4
TABLE 3:
(Continued) Study characteristics and results (only the significant data included)

With regard to methodological quality, 2 reviewers (E.M.P., B.P.) initially agreed on 84.1% of the items of the score lists. Disagreements were resolved through consensus. Table 1 shows the methodological quality, whereas Table 2 shows the reporting quality of the included studies. Table 3 displays the methods of the included studies.

The 8 studies included comprise a total of 654 children with constipation and 1931 with (constipation-associated) FI defined by different diagnostic criteria. Sex distribution was almost equal in children with constipation, whereas in children with FNRFI the majority was boys (11–18). The prevalence of constipation-associated FI and nonretentive FI varied greatly in the studies on FI (12,15–18). Details are presented in Table 3.

Exposure to Stressful Life Events

Table 3 depicts the several types of stressful life events as presented in the different studies and their level of significance. All studies on constipation and FI reported an association with stressful life events. The prevalence of stressful life events within the group of children with constipation varied from 1.6% to 82.2% (11,13,14). In children with FI, the prevalence of stressful life events varied from 44% to 90.9% (12,16). A level of significance was not given by Roma-Giannikou et al (14) and Levine (16), whereas Joinson et al (15) and Fishman et al (17) did not report an overall prevalence of stressful life events.

Out of 8 included studies, only 1 study measured the prevalence of child (sexual) abuse. van der Wal et al (18) showed a significantly higher prevalence of child (sexual) abuse in children with FI compared with healthy children with 0.7% of children with FI. No data have been reported on the nature of the abuse.

School-Related Stressful Life Events

As shown in Table 3, 4 of the included studies (12,13,15,16) assessed the impact of school-related stressful events. Being bullied at school remained significantly associated with FI after multiple logistic regression analysis in the Sri Lankan study on FI (12). In the study of Joinson et al (15), being the bully and being a relational victim were significantly associated with FI less than once a week, whereas being bullied did not remain significantly associated with FI after complete adjustment. None of the described determinants were significantly associated with FI once a week or more, except from being the bully unadjusted and adjusted for developmental delay. Separation from best friend and failure in an examination remained significant after multiple logistic regression analysis in the study of constipated Sri Lankan children (13). The study of Levine showed that 15% of the children with encopresis had a traumatic entry into school (16).

Family-Related Stressful Life Events

In the Sri Lankan studies, several family-related stressful life events were significantly related with functional defecation disorders, but none of those in the study on FI remained significant after multiple logistic regression analysis (12). In contrast, severe illness in a close family member, loss of job by a parent, and frequent punishment by parents remained significantly more common after multiple logistic regression analysis in constipated children (13). The study of Fishman et al (17) showed that interruption of toilet training and frequent punishment of parents during toilet training were significantly more common in children with primary FI than in children with secondary FI, whereas the study of Levine showed that family-related stressful life events were more common in children with secondary FI than in children with primary FI (16). Table 2 depicts the results on family-related stressful life events.

Other Stressful Life Events

Studies performed in Sri Lanka measured the effect of hospitalization and living in a war-affected area (see Table 2). Hospitalization showed to be a good predictor of FI, whereas hospitalization in the child with constipation did not (12,13). Living in a war-affected area was associated with constipation and remained significant after multiple logistic regression analysis (13).

DISCUSSION

This systematic review shows that the overall prevalence of stressful life events is significantly higher in children with functional defecation disorders compared with controls. The difference in prevalence of stressful life events between children with constipation and FI (constipation related FI or FNRFI) is negligible. Only 1 study in this systematic review reported a significantly higher prevalence of child (sexual) abuse in children with FI compared with controls, but clearly more data are needed to confirm this finding.

Stressful life events are common in childhood. In the Netherlands, 19% of the children are being bullied at least a few times a month, corresponding with the reported prevalence of bullying in other Western countries (47,48). It is known that victims of bullying have a significantly higher chance of developing new psychosomatic problems, such as functional abdominal pain and bedwetting (48).

Gilbert et al (49) reported that every year, 4% to 16% of children are physically abused and approximately 10% of children are neglected or psychologically abused. Furthermore, 15% to 30% of girls and 5% to 15% of boys are exposed to any type of sexual abuse during childhood. These data were gathered from population-based studies in developed countries (eg, Australia, United Kingdom, United States, Canada, New Zealand, Finland, Italy, Portugal). In the Netherlands, the second National Prevention Study of Child abuse found that approximately 34 per 1000 Dutch children had to deal with child abuse (50).

We can only speculate why stressful life events may lead to functional gastrointestinal disorders. Studies in adults suggest 2 mechanisms: childhood traumatic events may predispose an individual to psychopathology, which would lead to exaggerated reactions to stress and manifest as unexplained gastrointestinal symptoms (7,51); or childhood traumatic events may act as a contributory factor toward the sensitization of intestinal visceral afferents with its consequences (7). Others have proposed that after the experience of for instance sexual abuse, children may for instance experience urinary tract or anal inflammation and pain, which in turn leads to urinary incontinence, fecal impaction, or FI (52).

Since a few years, brain-imaging using functional magnetic resonance imaging (fMRI) is being used to explore the activity of several brain regions during visceral stimulation. Stress or abuse or trauma is believed to increase the activation of the anterior midcingulate cortex. Remarkably, patients with irritable bowel syndrome are reported to have a greater activation of the midcingulate cortex and other regions on visceral stimuli than healthy subjects, which is associated with pain experience (53). This could be one of the pathophysiological mechanisms that predispose children who experienced stressful life events or (sexual) abuse to develop functional defecation disorders as well.

The present review has several limitations. A potential shortcoming of this systematic review is the variation in defining and diagnosing functional constipation and FI in the included studies. In 4 population-based cross-sectional studies (11–13,15), a physical examination, including a rectal examination, was not performed. The studies on FI varied greatly with regard to the prevalence of constipation-associated FI and nonretentive FI (12,15–18). The absence of a physical examination may have resulted in a smaller percentage of children who were experiencing constipation-associated FI. The study of Joinson et al (15) and Fishman et al (17) are solely based on parental reports. Also, this may have contributed to the small percentage of constipation-associated FI.

In 5 studies questionnaires were based on recalled data (11–14,17). Although there is a potential risk of underreporting, caused by various factors including shame, van der Plas et al (54) showed that recalled data can accurately be used in a daily clinical setting, but special attention is necessary for episodes of FI and the size of stool.

One of the included studies only reported on symptoms and treatment for functional defecation disorders in the first 2 years of life and disruptive events during toilet training. The results showed a significant association between interruption of toilet training and punishment during toilet training and primary FI. Because this was a retrospective cross-sectional study and no adjustment for confounders have been made, there is a potential risk of reporting bias (17).

All included studies are cross-sectional studies (11–15,18). Therefore, we were unable to determine whether the functional defecation disorder was the result of a stressful life event or that the presence of a functional defecation disorder caused stress or was followed by a certain stressful life event.

The prevalence of stressful life events varied widely from 1.6% in Greece to 90.9% in Sri Lanka. Most likely this wide difference is caused by the diversity in study populations and the different definitions used for stressful life events. For example, in the Greek study only death or separation of a family member was considered to be a stressful life event, whereas in the Sri Lankan studies a broad list of circumstances were considered as stressful life events. In addition, Greece is a prosperous country (at the time of this writing), whereas Sri Lanka is a third world country dealing with the Sri Lankan Civil War during the period of study, which contributed substantially to the prevalence of stressful life events. In the study of Devanarayana and Rajindrajith (13) 46.8% of children with constipation lived in a war-effected area.

Finally, the majority of the included studies showed to have a considerable risk of bias (Table 1). Moreover, 5 of the 8 studies failed to provide mean ages and/or sex distributions of the control populations, making comparisons difficult, and draw firm conclusions.

This systematic review shows a high prevalence of stressful life events in children with functional defecation disorders. The majority of studies, however, showed a considerable risk of bias, and studies investigating the prevalence of child abuse in children with functional defecation disorders are lacking. On the basis of this review and clinical experience in our specialized outpatient clinic, we recommend physicians to inquire the history of stressful life events, including abuse in every patient presenting with a functional (defecation) disorder. Future research should focus on studies with a prospective case-control design in which stressful life events including child (sexual) abuse are systematically investigated in children with functional defecation disorders and compared with children with other functional disorders and healthy controls. When the true prevalence of stressful life events including abuse in children with defecation disorders will become available, an improved diagnostic and therapeutic approach for these children is possible. In addition, possible therapeutic approaches when significant stressful life events or child abuse are suspected must be investigated with preferably randomized controlled trials.

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Keywords:

children; functional defecation disorders; prevalence; stressful life events; systematic review

Supplemental Digital Content

© 2015 by European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology,