A marble in the small bowel.
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Biomedical subjects
Publications and source records attributed to Marc A Benninga.
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Fecal incontinence, the loss of feces in the underwear after age 4 years, is a frustrating phenomenon for children and their parents. It is difficult to treat, presenting as a single symptom without any organic cause or signs of constipation. This review addresses the definition of functional nonretentive fecal incontinence and provides an overview of its epidemiology, pathophysiology, clinical features, diagnostic work-up and prognosis.
OBJECTIVE: To clarify the relationship between disordered defecation and non-neuropathic bladder-sphincter dysfunction (NNBSD) by comparing the prevalence of symptoms of disordered defecation in children with NNBSD before and after treatment for urinary incontinence (UI), and assessing the effect of such symptoms on the cure rate for UI. PATIENTS AND METHODS: In the European Bladder Dysfunction Study, a prospective multicentre study comparing treatment plans for children with NNBSD, 202 children completed questionnaires on voiding and on defecation, at entry and after treatment for UI. Four symptoms of disordered defecation were evaluated; low defecation frequency, painful defecation, fecal soiling, and encopresis. RESULTS: At entry, 17 of the 179 children with complete data sets had low defecation frequency and/or painful defecation (9%), classified as functional constipation (FC). Of the 179 children, 57 had either isolated fecal soiling or soiling with encopresis (32%), classified as functional fecal incontinence (FFI). After treatment for UI, FFI decreased to 38/179 (21%) (statistically significant, P = 0.035); for FC there were too few children for analysis. After treatment for UI, 19 of the 179 children (11%) reported de novo FFI. Symptoms of disordered defecation did not influence the cure rate of treatment for UI. CONCLUSIONS: FFI improved significantly after treatment for UI only, but not in relation to the outcome of such treatment. FFI did not influence the cure rate for UI. There was little to support a causal relation between disordered defecation and NNBDS ('functional elimination syndrome').
Functional gastrointestinal disorder is a common problem in childhood. The symptoms vary from a relative mild gastrointestinal problem such as abdominal pain or infrequent defecation to severe problems with fecal impaction and fecal incontinence. The aim of this review is to describe and evaluate the value of the different existing methods to assess fecal loading on an abdominal radiograph with or without the use of radio-opaque markers in the diagnosis of functional abdominal pain, functional constipation and functional non-retentive fecal incontinence. In our opinion, the abdominal radiograph has limited value in the diagnostic work-up of children with functional gastrointestinal disorders.
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Children and adolescents with chronic defecation disorders and chronic abdominal pain without obvious organic etiology form a challenging group of patients for pediatric health-care professionals. The pathophysiologic mechanisms underlying such functional gastroenterology disorders are poorly understood. Research studies on the use of the barostat have been aimed to increase our knowledge in this area. Barostat testing allows defining visceral hyper- or hyposensitivity, contractility, and compliance of the gut. This review focuses on rectal barostat studies performed in children with abdominal pain, constipation, and fecal incontinence.
BACKGROUND & AIMS: Functional nonretentive fecal incontinence (FNRFI), incontinence in the absence of signs of fecal retention, is a frustrating phenomenon in children. No data on long-term outcome are available. The aim was to investigate the long-term outcome of FNRFI patients after intensive medical treatment. METHODS: Between 1990 and 1999, 119 patients (96 boys) with FNRFI were enrolled in 2 prospective, randomized trials investigating the effect of biofeedback training and/or laxative treatment. Follow-up (FU) was performed at 6 months, 1 year, and thereafter annually until September 2004. A standardized questionnaire was used to evaluate symptoms. Success was defined as a fecal incontinence frequency <1 per 2 weeks. RESULTS: Median age (25th-75th percentiles) was 9.2 years (range, 7.9-11.6 years). A 90% FU was achieved at all stages of the study. After 2 years of intensive therapy, 33 of 112 (29.5%) patients were successfully treated. The cumulative success percentage after 7 years of FU was 80%. At the biologic ages of 12 and 18 years, 49.4% (40/81) and 15.5% (9/58), respectively, of the patients still had fecal incontinence. Duration of fecal incontinence, with 4 years of age as the starting age for fecal incontinence (when a child should be toilet trained), was not related to successful outcome or relapse. Relapse occurred in 37% of patients. CONCLUSIONS: Only 29% of the patients with FNRFI were successfully treated after 2 years of intensive treatment. Despite recovery in the majority of patients beyond puberty, at age 18 years, 15% continued to have fecal incontinence.
OBJECTIVE: To evaluate rectal sensitivity in patients with pediatric constipation (PC) and nonretentive fecal soiling (FNRFS) using pressure-controlled distention (barostat). STUDY DESIGN: Thresholds for rectal sensitivity (first sensation, urge to defecate, and pain), and rectal compliance were determined using a barostat. RESULTS: A total of 69 patients with PC (50 males; mean age, 10.9 +/- 2.2 years) and 19 patients with FNRFS (15 males; mean age, 10.0 +/- 1.9 years) were compared with 22 healthy volunteers (HVs) (11 males; mean age, 12.7 +/- 2.6 years). Sensitivity thresholds were not significantly different among the 3 groups. Rectal compliance was increased in 58% of the patients with PC (P < .0001 vs HVs). Rectal compliance did not differ between patients with FNRFS and HVs. Children with PC with abnormal rectal function required significantly larger rectal volumes at urge to defecate. CONCLUSIONS: Increased compliance is the most prominent feature in patients with PC. Because of higher compliance in these children, larger stool volumes are required to reach the intrarectal pressure of the urge to defecate. Children with FNRFS have normal rectal function.
OBJECTIVE: To evaluate the effect of baclofen, a gamma-amino-butyric-acid B receptor agonist that inhibits transient lower esophageal sphincter relaxation (TLESR), on the rates of TLESR, gastroesophageal reflux (GER), and gastric emptying (GE) in children with GER disease. STUDY DESIGN: The efficacy of 0.5 mg/kg baclofen was evaluated in a randomized, double-blinded, placebo-controlled trial in 30 children. Patients were intubated with a manometric/pH assembly and given 250 mL of cow's milk. Esophageal motility and pH were then measured for 2 hours (control period). Baclofen or placebo was then administered, and 1 hour later 250 mL of milk was given again and measurements performed for another 2 hours (test period). The GE rate was measured by the (13)C octanoate breath test. RESULTS: Baclofen significantly reduced the incidence of TLESR (mean, 7.3 +/- 1.5 vs 3.6 +/- 1.2 TLESR/2 hours; P < .05) and acid GER (mean 4.2 +/- 0.7 vs 1.7 +/- 1.0 TLESR + GER/2 hours; P < .05) during the test period compared with the control period. Baclofen significantly accelerated the GE rate (median [interquartile range], GE(t1/2), 61 minutes [39, 81 minutes] vs 114 minutes [67, 170 minutes]; P < .05). Baclofen had no effect on the swallowing rate, pattern of esophageal peristalsis, or lower esophageal sphincter pressure. CONCLUSIONS: Baclofen reduces GER in children by inhibiting the triggering of TLESR. Baclofen also accelerates GE.
PURPOSE: The aim of this study was to define the predictive value of colonic manometry and contrast enema before cecostomy placement in children with defecation disorders. METHODS: Medical records, contrast enema, and colonic manometry studies were reviewed for 32 children with defecation disorders who underwent cecostomy placement between 1999 and 2004. Diagnoses included idiopathic constipation (n = 13), Hirschsprung's disease (n = 2), cerebral palsy (n = 1), imperforate anus (n = 6), spinal abnormality (n = 6), and anal with spinal abnormality (n = 4). Contrast enemas were evaluated for the presence of anatomic abnormalities and the degree of colonic dilatation. Colonic manometry was considered normal when high-amplitude propagating contractions (HAPC) occurred from proximal to distal colon. Clinical success was defined as normal defecation frequency with no or occasional fecal incontinence. RESULTS: Colonic manometry was done on 32 and contrast enema on 24 patients before cecostomy. At follow-up, 25 patients (78%) fulfilled the success criteria. Absence of HAPC throughout the colon was related to unsuccessful outcome (P = .03). Colonic response with normal HAPC after bisacodyl administration was predictive of success (P = .03). Presence of colonic dilatation was not associated with colonic dysmotility. CONCLUSION: Colonic manometry is helpful in predicting the outcome after cecostomy. Patients with generalized colonic dysmotility are less likely to benefit from use of antegrade enemas via cecostomy. Normal colonic response to bisacodyl predicts favorable outcome.
Recognizing the importance of childhood functional gastrointestinal disorders in understanding adult functional gastrointestinal disorders, and encouraging clinical and research interest, the Rome Coordinating Committee added a pediatric working team to Rome II in 1999. For Rome III, there was an increase from 1 to 2 pediatric working teams. This report summarizes the current consensus concerning functional disorders in infants and toddlers. Another report covers disorders diagnosed more often in school-aged children and adolescents. The symptoms from functional gastrointestinal disorders in children younger than 5 years depend on maturational factors in anatomy, gastrointestinal physiology, and intellectual and affective functioning. There has been little or no change for infant regurgitation, infant rumination syndrome, or infant dyschezia. Cyclic vomiting syndrome may be diagnosed after 2 rather than 3 episodes. The description of infant colic has been expanded, although there was consensus that infant colic does not reflect gastrointestinal malfunction. The greatest change was in functional constipation. Functional constipation and functional fecal retention in the 1999 report were merged into a single entity: functional constipation. Data-driven changes in diagnostic criteria for functional constipation appear to be less rigid and more inclusive than previous criteria.
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BACKGROUND: The data concerning the value of a plain abdominal radiograph in childhood constipation are inconsistent. Recently, positive results have been reported of a new radiographic scoring system, "the Leech method", for assessing faecal loading. OBJECTIVE: To assess intra- and interobserver variability and determine diagnostic accuracy of the Leech method in identifying children with functional constipation (FC). MATERIALS AND METHODS: A total of 89 children (median age 9.8 years) with functional gastrointestinal disorders were included in the study. Based on clinical parameters, 52 fulfilled the criteria for FC, six fulfilled the criteria for functional abdominal pain (FAP), and 31 for functional non-retentive faecal incontinence (FNRFI); the latter two groups provided the controls. To assess intra- and interobserver variability of the Leech method three scorers scored the same abdominal radiograph twice. A Leech score of 9 or more was considered as suggestive of constipation. ROC analysis was used to determine the diagnostic accuracy of the Leech method in separating patients with FC from control patients. RESULTS: Significant intraobserver variability was found between two scorers (P=0.005 and P<0.0001), whereas there was no systematic difference between the two scores of the other scorer (P=0.89). The scores between scorers differed systematically and displayed large variability. The area under the ROC curve was 0.68 (95% CI 0.58-0.80), indicating poor diagnostic accuracy. CONCLUSIONS: The Leech scoring method for assessing faecal loading on a plain abdominal radiograph is of limited value in the diagnosis of FC in children.
BACKGROUND: In children with severe generalized cerebral palsy, pneumonias are a major health issue. Malnutrition, dysphagia, gastro-oesophageal reflux, impaired respiratory function and constipation are hypothesized risk factors. Still, no data are available on the relative contribution of these possible risk factors in the described population. This paper describes the initiation of a study in 194 children with severe generalized cerebral palsy, on the prevalence and on the impact of these hypothesized risk factors of recurrent pneumonias. METHODS/DESIGN: A nested case-control design with 18 months follow-up was chosen. Dysphagia, respiratory function and constipation will be assessed at baseline, malnutrition and gastro-oesophageal reflux at the end of the follow-up. The study population consists of a representative population sample of children with severe generalized cerebral palsy. Inclusion was done through care-centres in a predefined geographical area and not through hospitals. All measurements will be done on-site which sets high demands on all measurements. If these demands were not met in "gold standard" methods, other methods were chosen. Although the inclusion period was prolonged, the desired sample size of 300 children was not met. With a consent rate of 33%, nearly 10% of all eligible children in The Netherlands are included (n = 194). The study population is subtly different from the non-participants with regard to severity of dysphagia and prevalence rates of pneumonias and gastro-oesophageal reflux. DISCUSSION: Ethical issues complicated the study design. Assessment of malnutrition and gastro-oesophageal reflux at baseline was considered unethical, since these conditions can be easily treated. Therefore, we postponed these diagnostics until the end of the follow-up. In order to include a representative sample, all eligible children in a predefined geographical area had to be contacted. To increase the consent rate, on-site measurements are of first choice, but timely inclusion is jeopardized. The initiation of this first study among children with severe neurological impairment led to specific, unexpected problems. Despite small differences between participants and non-participating children, our sample is as representative as can be expected from any population-based study and will provide important, new information to bring us further towards effective interventions to prevent pneumonias in this population.
BACKGROUND: Constipation is a common problem in children. Diagnosis is based on clinical features. In case of doubt about the presence of constipation, the existence of fecal retention can be evaluated on plain abdominal radiography. OBJECTIVES: To describe and to assess the evidence from observational, controlled studies concerning the association between abdominal radiography and symptoms and signs related to constipation in children. METHODS: MEDLINE was searched from inception to April 2004 using a specified search strategy. Studies that fulfilled predefined criteria were assessed for methodological quality. Study characteristics and associations were extracted and the results were summarized according to a best-evidence synthesis. RESULTS: Of the 392 publications identified, 6 studies met the inclusion criteria. Only 2 studies were of high methodological quality. The best-evidence synthesis yielded conflicting evidence for an association between a clinical and a radiological diagnosis of constipation. The likelihood ratio (LR) in 2 high-quality studies was close to 1 (LR, 1.2; 95% confidence interval [CI], 1.0-1.4; and LR, 1.0; 95% CI, 0.5-1.6). Conflicting evidence was found for an association between digital rectal examination and fecal impaction on radiography. Limited evidence was found for an association between a history of hard stool and a finding of rebound tenderness and radiography (LR, 1.2; 95% CI, 1.0-1.4; and LR, 1.1; 95% CI, 1.0-1.2, respectively). CONCLUSIONS: The limited amount of data available shows conflicting evidence for an association between clinical symptoms of constipation and fecal loading on abdominal radiographs in children. The recommendation to perform a plain abdominal radiograph in case of doubt of the presence of constipation in a child cannot be supported. Further research of good methodological quality is needed.
OBJECTIVE: To compare the diagnostic accuracy of contrast enema (CE), anorectal manometry (ARM), and rectal suction biopsy (RSB) for the detection of Hirschsprung's disease (HD). STUDY DESIGN: Following a prospective protocol, infants suspected of HD underwent all 3 index tests. Children with positive results on 2 or more index tests or who continued to have severe bowel problems underwent a full thickness biopsy as reference standard. Clinical follow-up was the reference standard in all other children. RESULTS: Between 2000 and 2003, 111 consecutive patients (67 boys; median age, 5.3 months) in whom HD was suspected were enrolled. HD was found in 28 patients. RSB had the highest sensitivity (93%) and specificity (100%) rates, but values were not significantly different from CE (sensitivity, 76%; specificity, 97%) or from ARM (sensitivity, 83%; specificity, 93%). Inconclusive test results occurred in 8 infants with CE, in 15 infants with ARM because of agitation, and in 2 infants with RSB. CONCLUSION: RSB is the most accurate test for diagnosing HD, and it has the lowest rate of inconclusive test results.
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