Colonic hyperactivity results in frequent fecal soiling in a subset of children after surgery for Hirschsprung disease
ABSTRACT Fecal soiling is a challenging problem in some children after pull-through surgery for Hirschsprung disease (HSCR). The prevailing perception is that soiling results from overflow incontinence; however, its treatment with laxatives yields mixed results. Colonic manometry studies are reported to be normal in most patients in this population. The interpretation of these findings does not support the physiology of fecal overflow incontinence in these children. The aim of the present study was to define the physiology underlying daily, frequent fecal soiling in children after surgery for HSCR using manometric techniques.
Four pediatric motility centers in the United States participated in the study; medical records and manometric tracings (anorectal and colonic) of children (n = 59; 6.5 years; 48 boys) who had pull-through surgery for HSCR and presented with daily, frequent fecal soiling were examined. Children referred for evaluation of constipation who had normal colonic manometry served as controls (n = 25; 6.7 years; 12 boys). The patients with HSCR were divided into 2 groups (Hirschsprung disease groups 1 and 2 [HD1, HD2]) based on the absence or presence of high-amplitude propagated contractions (HAPCs). A control group that included children with chronic constipation was also studied. We compared the mean HAPC frequency between the HD2 and control groups.
HD1 included 21 patients who had no HAPCs in fasting or postprandial periods. HD2 included 38 patients who had an average of 0.07 HPACs/min while fasting and 0.13/min in the postprandial state. In this subset the number of HAPCs in the fasting state (P = 0.04) and the postprandial state (P < 0.001) was greater when compared with controls. Additionally, there was a significant increase in HAPCs/min from the fasting to the postprandial state (P = 0.01). In the HD2 group 40% had colonic hyperactivity.
Daily, frequent fecal soiling after pull-through surgery for HSCR may be due to colonic hyperactivity in some children. It is imperative that this unique subset be identified because the management strategy would include avoidance of laxatives, contrary to standard current practice.
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ABSTRACT: The main objective of this study is to evaluate the efficacy of pelvic floor muscle exercise for the treatment of fecal incontinence after Soave procedure for Hirschsprung disease (HD). A case series study was performed in 24 incontinent children after Soave pull-through surgery for HD. Out of the 24 patients, 16 patients (training group) received pelvic floor muscle training while the other 8 patients (control group) did not receive further treatment. For children who received pelvic floor muscle training, biofeedback treatment was given for 2 weeks in hospital and they were then instructed to carry out pelvic floor muscle exercise at home. At the baseline and after 1 year of training, anorectal manometry was performed to measure resting anal canal pressure, squeeze pressure, and rectal sensation. Efficacy of pelvic floor muscle exercise for the treatment of postoperative fecal incontinence was evaluated by the difference between baseline and 1-year follow-up values. At the baseline, the characteristics of the incontinent children were also compared with 18 children who were performed Soave operation for HD and had normal anal function. Lower resting anal canal pressure distinguished the incontinent children from the continent ones. Resting pressure of the incontinent children was significantly improved by pelvic floor muscle exercise: the baseline and 1-year follow-up values of the treatment group were 18.6 ± 6.2 and 35.4 ± 8.7 mm Hg, respectively. Squeeze pressure and clinical outcomes were also improved after the pelvic floor muscle training: only 3 out of the 16 patients had occasional soiling after the training. No significant changes in clinical outcome and manometry measurements were observed in the control group. The damage of internal anal sphincter might be one of the causes of fecal incontinence after Soave procedure. The damage of internal anal sphincter could be caused by lower level of dissection, vigorous anal dilation, and excessive anal canal traction during operation. Pelvic floor muscle training is one procedure of choice to treat this complaint.European Journal of Pediatric Surgery 05/2012; 22(4):300-4. DOI:10.1055/s-0032-1313351 · 0.98 Impact Factor
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ABSTRACT: Background Antegrade continence enemas (ACE) have been used in the treatment of defecation disorders in children; little is known on their effect on colon motility and the utility of the colon manometry (CM) predicting long-term ACE outcomes. Methods Retrospective review of children with constipation undergoing CM before and after ACE to evaluate CM changes and their utility on predicting ACE outcome. Key Results A total of 40 patients (mean age 8.8 SD 3 years and 53% female patients) were included; 39 of 40 responded to the ACE. Of these 39, 14 (36%) were dependent and 25 (64%) had decreased it (11 of those or 28% discontinued it). On repeat CM we found a significant increase in the fasting (P < 0.01) and postprandial (P = 0.03) motility index, number of bisacodyl-induced high amplitude propagating contractions (HAPCs) (P = 0.03), and total HAPCs (P = 0.02). Gastrocolonic response to a meal, propagation and normalization of HAPCs improved in 28%, 58%, and 33%, respectively, with CM normalizing in 33% of patients. The baseline CM did not predict ACE outcome. The presence of normal HAPCs on the repeat CM was associated with ACE decrease. Progression and normalization of HAPCs (P = 0.01 and 0.02, respectively) and CM normalization (P = 0.01) on repeat CM were individually associated with ACE decrease. No CM change was associated with ACE discontinuation. Multivariate analysis showed that older age and HAPC normalization on CM predict ACE decrease and older age is the only predictor for ACE discontinuation. Conclusions & Inferences Colon motility improves after ACE and the changes on the repeat CM may assist in predicting ACE outcome.Neurogastroenterology and Motility 10/2012; 25(2). DOI:10.1111/nmo.12018 · 3.42 Impact Factor
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ABSTRACT: Despite significant developments in the understanding of the pathologic anatomy and physiology of Hirschsprung's disease (HD), the results of surgical therapy remain far from perfect. The functional defects and psychosocial difficulties that occur commonly in children with HD are passed on to adulthood in a significant proportion of patients. Recent prospective and adequately controlled cross-sectional studies reveal that constipation and fecal soiling are common late sequelae in adulthood. HD patients show uniformly lower scores of overall bowel function than healthy control subjects. Functional outcome and quality of life may be interrelated and deteriorate with increasing age. Despite these shortcomings, at adult age, most of the HD patients appear to be able to function as normal members of the society in terms of psychosocial, occupational, and recreational activities. Patients are at risk of developing related conditions, such as cancer, that require planning of specific screening programs. Many of the long-term problems that these patients experience are specific to HD. The follow-up of HD patients should be performed by medical personnel familiar with the disease, preferably in a specialized referral center, and the follow-up should continue beyond childhood.Seminars in Pediatric Surgery 11/2012; 21(4):336-43. DOI:10.1053/j.sempedsurg.2012.07.008 · 1.94 Impact Factor