INVITED REVIEW
Stressful Life Events in Children With Functional Defecation Disorders
Elise M. Philips, Babette Peeters, yArianne H. Teeuw, zArnold G.E. Leenders, §Nicole Boluyt, y Sonja N. Brilleslijper-Kater, and Marc A. Benninga
ABSTRACT Objectives: The aim of the study was to determine the prevalence of stressful life events including (sexual) abuse in children with functional defecation disorders by performing a systematic review. Methods: We searched MEDLINE, EMBASE, and PsycINFO for cohort, case-control and cross-sectional studies investigating the prevalence of stressful life events, including (sexual) abuse in children with functional defecation disorders. Results: The search yielded 946 articles, of which 8 were included with data from 654 children with functional constipation and 1931 children with (constipation-associated) fecal incontinence (FI). Overall, children with functional defecation disorders had been significantly more exposed to stressful life events than healthy children, with prevalence rates ranging from 1.6% to 90.9%. Being bullied, being a relational victim, interruption of toilet training, punishment by parents during toilet training, and hospitalization were significantly related to FI, whereas separation from the best friend, failure in an examination, severe illness in a close family member, loss of job by a parent, frequent punishment, and living in a war-affected area were significantly related to constipation. Only 1 study measured the prevalence of child abuse, which reported a significantly higher prevalence of child (sexual) abuse in children with FI compared with controls. Conclusions: The prevalence of stressful life events, including (sexual) abuse is significantly higher in children with functional defecation disorders compared with healthy children. To gain more insight into the true prevalence of child (sexual) abuse in children with functional defecation disorders, more studies are clearly needed. Key Words: children, functional defecation disorders, prevalence, stressful life events, systematic review
(JPGN 2015;61: 384–392)
Received November 12, 2014; accepted June 5, 2015. From the Department of Pediatric Gastroenterology and Nutrition, the yDepartment of Social Pediatrics, Emma Children’s Hospital, Academic Medical Center, the zMedical Library, Academic Medical Center Amsterdam, and the §Department of Pediatrics, Emma Children’s Hospital, Academic Medical Center, Amsterdam, The Netherlands. Address correspondence and reprint requests to Elise Philips, MD, Department of Pediatric Gastroenterology and Nutrition, Emma Children’s Hospital, Academic Medical Center, Room C2–312, Meibergdreef 9, 1105 AZ Amsterdam, The Netherlands (e-mail: [email protected]). Supplemental digital content is available for this article. Direct URL citations appear in the printed text, and links to the digital files are provided in the HTML text of this article on the journal’s Web site (www.jpgn.org). The authors report no conflicts of interest. Copyright # 2015 by European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition DOI: 10.1097/MPG.0000000000000882
C
onstipation 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, http:// 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.
384 JPGN Volume 61, Number 4, October 2015 Copyright 2015 by ESPGHAN and NASPGHAN. Unauthorized reproduction of this article is prohibited.
JPGN
Volume 61, Number 4, October 2015
Stressful Life Events in Defecation Disorders
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.
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 (þþ), minor (þ), 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.
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.
Quality Assessment
RESULTS
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
The search generated 946 titles and abstracts (Supplemental Figure 1, Supplemental Digital Content 2, http://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
TABLE 1. Guidelines and checklist for appraising a medical article according to Fowkes and Fulton (8) Rajindrajith Devanarayana Inan Joinson Van der Wal Fishman Roma-Giannikou et al (12) and Rajindrajith (13) et al (11) et al (15) et al (18) et al (17) et al (14) 1. Study design appropriate to objectives? 2. Study sample representative? Source of sample Sampling method Sample size Entry criteria/exclusions Nonrespondents 3. Control group acceptable? Definition of controls Source of controls Matching/randomization Comparable characteristics 4. Quality of measurements and outcomes? Validity Reproducibility Blindness Quality control 5. Completeness? Compliance Dropouts Deaths Missing data 6. Distorting influences? Extraneous treatments Contamination Changes over time Confounding factors Distortion reduced by analysis
þ þ þ 0 0 þ Unknown 0 0 0 NA þ þ þ þ NA þ 0 NA NA NA 0 þ NA NA NA þþ 0
þ þ þ 0 0 þ Unknown 0 0 0 NA 0 þ þ þ NA þ Unknown NA NA NA Unknown þ NA NA NA þþ 0
þ þ þ 0 0 þ Unknown 0 0 0 NA 0 þ þ þ NA þ Unknown NA NA NA Unknown þ NA NA NA þþ 0
þ 0 0 0 0 þ 0 0 0 0 NA þ 0 0 0 NA 0 0 NA NA NA 0 0 NA NA NA 0 0
þ 0 0 0 0 þ 0 Unknown Unknown 0 NA Unknown 0 0 0 þ 0 Unknown NA NA NA Unknown þ NA NA NA þþ 0
þ 0 þ 0 0 0 0 NA NA NA NA NA 0 0 0 NA þ Unknown NA NA NA Unknown þ NA NA NA þþ 0
þ 0 0 0 0 0 Unknown Unknown 0 0 0 Unknown þ 0 0 þ þ Unknown NA NA NA Unknown þ NA NA NA þþ 0
Levine (16) þ 0 þ 0 0 0 0 NA NA NA NA NA þ þ þ NA þ Unknown NA NA NA Unknown þ NA NA NA þþ 0
Each criterion has been assigned as major (þþ), minor (þ), or no problem (0) in terms of their expected effect on the results. Not applicable (NA) has been used when not applicable and Unknown when a criterion is unknown.
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TABLE 2. Reporting quality of the included studies Devanarayana and Rajindrajith et al (12) Rajindrajith (13) Study design 1. Study design clearly described Setting 2. Description of the setting, locations, and relevant dates, including periods of recruitment, follow-up, and data collection Study population 3. Description of eligibility criteria, and the sources and methods of selection of participants 4. Matching control group 5. Description of study populations At least mentioned are age; sex; duration of constipation; and severity of constipation (defecation frequency or presence of FI) 6. Description of inclusion and exclusion criteria Variables/outcome measures 7. Clear definition of outcome measures 8. Description of possible confounders Bias 9. Potential sources of bias are addressed Study size 10. Explanation of the study size Statistical methods 11. Description of all statistical methods, including those used to control for confounding, and how missing data were addressed
Inan et al (11)
Joinson et al (15)
Van der Wal et al (18)
Fishman et al (17)
Roma-Giannikou Levine et al (14) (16)
Yes
Yes
Yes
Yes
Yes
Yes
Yes
Yes
No
Yes
Yes
Yes
Yes
Yes
No
Yes
Yes
No
Yes
Yes
Yes
Yes
Yes
No
No No
Do not know No
Do not know Do not know Do not know Do not know No No No No
Do not know No
No No
No
No
No
No
No
Yes
Yes
Yes
No No
Yes No
Yes No
Yes Yes
Yes No
Yes No
Yes No
No No
No
No
No
Yes
Yes
Yes
No
No
No
No
No
No
No
No
No
No
No
No
No
Yes
No
No
No
No
FI ¼ fecal incontinence.
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, http://links.lww.com/MPG/A511). 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
386
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 www.jpgn.org
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Cross-sectional
Cross-sectional
Cross-sectional
Cross-sectional
Cross-sectional
Cross-sectional
Cross-sectional
Cross-sectional
Rajindrajith et al (12)
Devanarayana et al (13)
Inan et al (11)
Joinson et al (15)
Van der Wal et al (18)
Fishman et al (17)
Roma-Giannikou et al (14)
Levine (16)
1932 children ages 2–14 y, mean, age and boy-to-girl ratio not presented 102 children ages 4–13 y (7.33, SD n/a) 85.3% male
Pediatric outpatient clinic in Boston, MA. Interviews and questionnaires. Physical including rectal examination performed. No statistical analysis performed
A random stratified sample from 3 counties of Greece. Interviews from parents and children. Physical examination of constipated children, including anal and rectal examination. Data were analyzed using x2 tests.
Children referred to the Pains and Incontinence Program at Children’s Hospital in Boston. Detailed parent questionnaire. Full medical history and physical examination performed. Parametric variables were compared using the t test, and categorical variables were compared using x2 tests.
Children attended school health care centers for routine medical checks in Amsterdam, The Netherlands. Parent questionnaire filled out by school doctors, child health care findings and action register. Data were analyzed using x2 tests. A logistical regression was performed to determine the association of each sociodemographic characteristic to the prevalence of encopresis. Adjustments were made for the effect of confounding variables, including child’s age, sex, the number of children living at home, ethnic origin of the family, and the postal code.
School-based, island-wide study in Sri Lanka. Random selection of provinces, schools, and classes. Validated, self-administered questionnaire. No (rectal) examinations performed. Data were analyzed using x2 and Fisher exact tests. Multiple logistic regression analysis was performed on variables that were found to have significant association. School-based, island-wide study in Sri Lanka. Random selection of provinces and schools. Validated, self-administered questionnaire. No physical examination performed. Data were analyzed using x2 and Fisher exact tests. Multiple logistic regression analysis was performed on variables that were found to have significant association. School-based population in Edirne, Turkey. Randomly selected from class lists. Self-administered questionnaire by parents and children. No physical examination performed. Data were analyzed using x2 and Fisher exact tests. To determine the potential risk factors of constipation, a logistic regression model was built. The Avon Longitudinal Study of Parents and Children population-based cohort. Interviews and questionnaires. No physical examination performed. Data were analyzed using x2 tests. A multivariable analysis was performed to determine which emotional and behavior problems were associated with soiling. Adjustments were made for the effect of confounding variables, including developmental delay, sex, stressful life events, and family sociodemographic background. Missing data were excluded from the multivariable models. In the final adjusted estimates, a missing data imputation technique was used known as ‘‘ice’’ to correct for any bias caused by dropout.
Methods Prevalence of constipationassociated and NRFI and associated factors
Association between constipation and stressful life events
Prevalence of constipation and associated factors
Prevalence of psychological, emotional, and behavioral problems, self-esteem, bullying, and friendships in children who soil compared with children who do not soil
Prevalence of encopresis and associated psychosocial problems
Prevalence of predisposing factors that occurred in the first 2 years of life and around toilet training associated with childhood encopresis
Prevalence of constipation, the relations to socioeconomic factors and clinical presentation
Rome III criteria/defecation into places inappropriate to the social context, at least once per month, for a minimum period of 2 months/Rome III criteria
Rome III criteria/–/–
NASPGHAN criteria/–/–
–/Repeated passage of feces in inappropriate places, with a frequency of at least once a month for 3 months in children age 4 years and in the absence of a physical cause/–
–/Involuntary loss of feces in the underwear once a month or more/–
Hard stools or difficulty in defecation sufficient to cause significant distress according to the parents’ perception/ Repeated passage of feces into inappropriate places (such as underwear or pajamas), whether involuntary or intentional, after the age of 4 years/– 3 or less bowel movements per week, or hard stools with painful defecation (even with more than 3 bowel movements per week) at least for the last 2 months/–/– –/Any child over 4 years of age who regularly passed (semi)formed or liquid stools into his underwear or pajamas with no apparent primary organic etiology/–
Encopresis-associated clusters of demographic, social, and training factors
Outcome measure
Definition of constipation/FI/(F)NRFI
102 children with FI (7.33, SD n/a), 85.3% male. 40 children with primary FI (6.25 y, SD n/a) 85.1% male 62 children with secondary FI (8.0 y, SD n/a) 85.5% male. 79.4% stool impaction at first visit
565 children with FI, mean age, and boy-to-girl ratio not presented 117 children with frequent FI (1/week), mean age not presented, 69% male 448 children with less frequent FI (
Stressful Life Events in Children With Functional Defecation Disorders
Elise M. Philips, Babette Peeters, yArianne H. Teeuw, zArnold G.E. Leenders, §Nicole Boluyt, y Sonja N. Brilleslijper-Kater, and Marc A. Benninga
ABSTRACT Objectives: The aim of the study was to determine the prevalence of stressful life events including (sexual) abuse in children with functional defecation disorders by performing a systematic review. Methods: We searched MEDLINE, EMBASE, and PsycINFO for cohort, case-control and cross-sectional studies investigating the prevalence of stressful life events, including (sexual) abuse in children with functional defecation disorders. Results: The search yielded 946 articles, of which 8 were included with data from 654 children with functional constipation and 1931 children with (constipation-associated) fecal incontinence (FI). Overall, children with functional defecation disorders had been significantly more exposed to stressful life events than healthy children, with prevalence rates ranging from 1.6% to 90.9%. Being bullied, being a relational victim, interruption of toilet training, punishment by parents during toilet training, and hospitalization were significantly related to FI, whereas separation from the best friend, failure in an examination, severe illness in a close family member, loss of job by a parent, frequent punishment, and living in a war-affected area were significantly related to constipation. Only 1 study measured the prevalence of child abuse, which reported a significantly higher prevalence of child (sexual) abuse in children with FI compared with controls. Conclusions: The prevalence of stressful life events, including (sexual) abuse is significantly higher in children with functional defecation disorders compared with healthy children. To gain more insight into the true prevalence of child (sexual) abuse in children with functional defecation disorders, more studies are clearly needed. Key Words: children, functional defecation disorders, prevalence, stressful life events, systematic review
(JPGN 2015;61: 384–392)
Received November 12, 2014; accepted June 5, 2015. From the Department of Pediatric Gastroenterology and Nutrition, the yDepartment of Social Pediatrics, Emma Children’s Hospital, Academic Medical Center, the zMedical Library, Academic Medical Center Amsterdam, and the §Department of Pediatrics, Emma Children’s Hospital, Academic Medical Center, Amsterdam, The Netherlands. Address correspondence and reprint requests to Elise Philips, MD, Department of Pediatric Gastroenterology and Nutrition, Emma Children’s Hospital, Academic Medical Center, Room C2–312, Meibergdreef 9, 1105 AZ Amsterdam, The Netherlands (e-mail: [email protected]). Supplemental digital content is available for this article. Direct URL citations appear in the printed text, and links to the digital files are provided in the HTML text of this article on the journal’s Web site (www.jpgn.org). The authors report no conflicts of interest. Copyright # 2015 by European Society for Pediatric Gastroenterology, Hepatology, and Nutrition and North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition DOI: 10.1097/MPG.0000000000000882
C
onstipation 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, http:// 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.
384 JPGN Volume 61, Number 4, October 2015 Copyright 2015 by ESPGHAN and NASPGHAN. Unauthorized reproduction of this article is prohibited.
JPGN
Volume 61, Number 4, October 2015
Stressful Life Events in Defecation Disorders
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.
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 (þþ), minor (þ), 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.
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.
Quality Assessment
RESULTS
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
The search generated 946 titles and abstracts (Supplemental Figure 1, Supplemental Digital Content 2, http://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
TABLE 1. Guidelines and checklist for appraising a medical article according to Fowkes and Fulton (8) Rajindrajith Devanarayana Inan Joinson Van der Wal Fishman Roma-Giannikou et al (12) and Rajindrajith (13) et al (11) et al (15) et al (18) et al (17) et al (14) 1. Study design appropriate to objectives? 2. Study sample representative? Source of sample Sampling method Sample size Entry criteria/exclusions Nonrespondents 3. Control group acceptable? Definition of controls Source of controls Matching/randomization Comparable characteristics 4. Quality of measurements and outcomes? Validity Reproducibility Blindness Quality control 5. Completeness? Compliance Dropouts Deaths Missing data 6. Distorting influences? Extraneous treatments Contamination Changes over time Confounding factors Distortion reduced by analysis
þ þ þ 0 0 þ Unknown 0 0 0 NA þ þ þ þ NA þ 0 NA NA NA 0 þ NA NA NA þþ 0
þ þ þ 0 0 þ Unknown 0 0 0 NA 0 þ þ þ NA þ Unknown NA NA NA Unknown þ NA NA NA þþ 0
þ þ þ 0 0 þ Unknown 0 0 0 NA 0 þ þ þ NA þ Unknown NA NA NA Unknown þ NA NA NA þþ 0
þ 0 0 0 0 þ 0 0 0 0 NA þ 0 0 0 NA 0 0 NA NA NA 0 0 NA NA NA 0 0
þ 0 0 0 0 þ 0 Unknown Unknown 0 NA Unknown 0 0 0 þ 0 Unknown NA NA NA Unknown þ NA NA NA þþ 0
þ 0 þ 0 0 0 0 NA NA NA NA NA 0 0 0 NA þ Unknown NA NA NA Unknown þ NA NA NA þþ 0
þ 0 0 0 0 0 Unknown Unknown 0 0 0 Unknown þ 0 0 þ þ Unknown NA NA NA Unknown þ NA NA NA þþ 0
Levine (16) þ 0 þ 0 0 0 0 NA NA NA NA NA þ þ þ NA þ Unknown NA NA NA Unknown þ NA NA NA þþ 0
Each criterion has been assigned as major (þþ), minor (þ), or no problem (0) in terms of their expected effect on the results. Not applicable (NA) has been used when not applicable and Unknown when a criterion is unknown.
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TABLE 2. Reporting quality of the included studies Devanarayana and Rajindrajith et al (12) Rajindrajith (13) Study design 1. Study design clearly described Setting 2. Description of the setting, locations, and relevant dates, including periods of recruitment, follow-up, and data collection Study population 3. Description of eligibility criteria, and the sources and methods of selection of participants 4. Matching control group 5. Description of study populations At least mentioned are age; sex; duration of constipation; and severity of constipation (defecation frequency or presence of FI) 6. Description of inclusion and exclusion criteria Variables/outcome measures 7. Clear definition of outcome measures 8. Description of possible confounders Bias 9. Potential sources of bias are addressed Study size 10. Explanation of the study size Statistical methods 11. Description of all statistical methods, including those used to control for confounding, and how missing data were addressed
Inan et al (11)
Joinson et al (15)
Van der Wal et al (18)
Fishman et al (17)
Roma-Giannikou Levine et al (14) (16)
Yes
Yes
Yes
Yes
Yes
Yes
Yes
Yes
No
Yes
Yes
Yes
Yes
Yes
No
Yes
Yes
No
Yes
Yes
Yes
Yes
Yes
No
No No
Do not know No
Do not know Do not know Do not know Do not know No No No No
Do not know No
No No
No
No
No
No
No
Yes
Yes
Yes
No No
Yes No
Yes No
Yes Yes
Yes No
Yes No
Yes No
No No
No
No
No
Yes
Yes
Yes
No
No
No
No
No
No
No
No
No
No
No
No
No
Yes
No
No
No
No
FI ¼ fecal incontinence.
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, http://links.lww.com/MPG/A511). 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
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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 www.jpgn.org
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Rajindrajith et al (12)
Devanarayana et al (13)
Inan et al (11)
Joinson et al (15)
Van der Wal et al (18)
Fishman et al (17)
Roma-Giannikou et al (14)
Levine (16)
1932 children ages 2–14 y, mean, age and boy-to-girl ratio not presented 102 children ages 4–13 y (7.33, SD n/a) 85.3% male
Pediatric outpatient clinic in Boston, MA. Interviews and questionnaires. Physical including rectal examination performed. No statistical analysis performed
A random stratified sample from 3 counties of Greece. Interviews from parents and children. Physical examination of constipated children, including anal and rectal examination. Data were analyzed using x2 tests.
Children referred to the Pains and Incontinence Program at Children’s Hospital in Boston. Detailed parent questionnaire. Full medical history and physical examination performed. Parametric variables were compared using the t test, and categorical variables were compared using x2 tests.
Children attended school health care centers for routine medical checks in Amsterdam, The Netherlands. Parent questionnaire filled out by school doctors, child health care findings and action register. Data were analyzed using x2 tests. A logistical regression was performed to determine the association of each sociodemographic characteristic to the prevalence of encopresis. Adjustments were made for the effect of confounding variables, including child’s age, sex, the number of children living at home, ethnic origin of the family, and the postal code.
School-based, island-wide study in Sri Lanka. Random selection of provinces, schools, and classes. Validated, self-administered questionnaire. No (rectal) examinations performed. Data were analyzed using x2 and Fisher exact tests. Multiple logistic regression analysis was performed on variables that were found to have significant association. School-based, island-wide study in Sri Lanka. Random selection of provinces and schools. Validated, self-administered questionnaire. No physical examination performed. Data were analyzed using x2 and Fisher exact tests. Multiple logistic regression analysis was performed on variables that were found to have significant association. School-based population in Edirne, Turkey. Randomly selected from class lists. Self-administered questionnaire by parents and children. No physical examination performed. Data were analyzed using x2 and Fisher exact tests. To determine the potential risk factors of constipation, a logistic regression model was built. The Avon Longitudinal Study of Parents and Children population-based cohort. Interviews and questionnaires. No physical examination performed. Data were analyzed using x2 tests. A multivariable analysis was performed to determine which emotional and behavior problems were associated with soiling. Adjustments were made for the effect of confounding variables, including developmental delay, sex, stressful life events, and family sociodemographic background. Missing data were excluded from the multivariable models. In the final adjusted estimates, a missing data imputation technique was used known as ‘‘ice’’ to correct for any bias caused by dropout.
Methods Prevalence of constipationassociated and NRFI and associated factors
Association between constipation and stressful life events
Prevalence of constipation and associated factors
Prevalence of psychological, emotional, and behavioral problems, self-esteem, bullying, and friendships in children who soil compared with children who do not soil
Prevalence of encopresis and associated psychosocial problems
Prevalence of predisposing factors that occurred in the first 2 years of life and around toilet training associated with childhood encopresis
Prevalence of constipation, the relations to socioeconomic factors and clinical presentation
Rome III criteria/defecation into places inappropriate to the social context, at least once per month, for a minimum period of 2 months/Rome III criteria
Rome III criteria/–/–
NASPGHAN criteria/–/–
–/Repeated passage of feces in inappropriate places, with a frequency of at least once a month for 3 months in children age 4 years and in the absence of a physical cause/–
–/Involuntary loss of feces in the underwear once a month or more/–
Hard stools or difficulty in defecation sufficient to cause significant distress according to the parents’ perception/ Repeated passage of feces into inappropriate places (such as underwear or pajamas), whether involuntary or intentional, after the age of 4 years/– 3 or less bowel movements per week, or hard stools with painful defecation (even with more than 3 bowel movements per week) at least for the last 2 months/–/– –/Any child over 4 years of age who regularly passed (semi)formed or liquid stools into his underwear or pajamas with no apparent primary organic etiology/–
Encopresis-associated clusters of demographic, social, and training factors
Outcome measure
Definition of constipation/FI/(F)NRFI
102 children with FI (7.33, SD n/a), 85.3% male. 40 children with primary FI (6.25 y, SD n/a) 85.1% male 62 children with secondary FI (8.0 y, SD n/a) 85.5% male. 79.4% stool impaction at first visit
565 children with FI, mean age, and boy-to-girl ratio not presented 117 children with frequent FI (1/week), mean age not presented, 69% male 448 children with less frequent FI (
