Background
After Hirschsprung’s disease (HD) surgery, many children suffer fecal incontinence caused by increased number of high amplitude propagating contractions (HAPCs) propagating through the neorectum to the anal verge. The aim of this study was to determine whether children with HD have more HAPCs than children with colon transections for reasons other than HD.
Methods
We reviewed 500 colon manometries. Children (7.6±5.1 yrs; 275 male) with functional constipation (n=237; 7.4±5.0yrs; 126 male) and chronic abdominal pain (n=48; 9.8±5.8yrs; 25 male) served as controls compared to subjects with HD (n=56; 6.9±4.1yrs; 44 male) and colon transection for other reasons (n=24; 6.1±5.8yrs; 12 male). We excluded 139 subjects without HAPCs. We documented HAPCs during 1 h fasting and 1 h postprandial. Results are mean ± SD.
Results
During fasting, HD subjects had more HAPCs (2.2±3.4/h) vs. functional constipation (0.8±2.2/h, p=0.0004) and chronic pain (0.5±1.1/h, p=0.001), but not more than colon transection (1.9±3.2/h, p=1.0). HD showed more postprandial HAPCs (4.0±5.4/h) than functional constipation (1.5±2.5/h, p<0.0001) and chronic pain (0.9±1.6/h, p<0.0001), but not more than colon transection (2.4±3.0/h, p=0.6). There were more HAPCs fasting and post-prandial after colon transection (1.9 ± 3.2/h and 2.4±3.0/h) than functional constipation (0.8±2.2/h, p=0.3 and 1.5±2.5/h, p=1.0) and chronic pain (0.5±1.1/h, p=1.0 and 0.9±1.6, p=1.0). HD subjects were divided by chief complaint: fecal incontinence or constipation. HD subjects with incontinence (n=23) only had more HAPCs fasting (p=0.01) and post-prandial (p=0.01) than HD subjects with constipation (n=28) only.
Conclusions
Increased HAPCs followed colon transection, regardless of cause. HD subjects with incontinence had more HAPCs than subjects with colon transection for other reasons.
Objectives: Four-hour gastric emptying scintigraphy (GES) is the recommended method to identify both adult and childhood gastroparesis (GP). Previous pediatric studies have, however, not used this standard. We sought to determine the characteristics and outcomes of children versus adolescents with GP using the 4-hour GES evaluation. Methods: We performed a retrospective chart review of pediatric patients diagnosed with GP by 4-hour GES (>10% retention at 4 hours). Demographics, body mass index, GP-related symptoms, comorbidities, etiologies, therapies (eg, medications), healthcare utilization, and response to therapy were captured systematically. Symptoms were compared from the initial versus last gastroenterology visit. Outcomes were categorized as no improvement; improvement (resolution of at least 1 symptom while remaining on therapy); and complete resolution of symptoms. Results: A total of 239 subjects (12.1 AE 4.1 years [mean AE standard deviation], 70% girls) were included. The identified characteristics of childhood GP were broad with idiopathic GP being the most common etiology. Outcomes over a median of 22 months (25%-75%: 9.0-45.5 months) were 34.8% no improvement, 34.8% some improvement, and 30.3% with complete symptom resolution. Compared to younger children, adolescents had a higher female predominance (P < 0.01) and were more likely to have nausea (P ¼ 0.006). Girls were more likely to have abdominal pain (P ¼ 0.001), nausea (P ¼ 0.03), and a documented diagnosis of dysautonomia (P ¼ 0.03). Boys were more likely to have regurgitation (P ¼ 0.006), gastroesophageal reflux disease (P ¼ 0.02), and rumination (P ¼ 0.02). Conclusions: Using the 4-hour GES standard, childhood GP has broad clinical characteristics and outcomes. There are several significant age-and sex-based differences in childhood GP.
Hirschsprung's disease (HSCR) is a congenital defect caused by impaired development of the enteric nervous system. Inflammatory bowel disease has an increased prevalence in patients with HSCR. We describe the clinical course of a patient with long-segment HSCR who, at the age of 12 months, developed diffuse intestinal inflammation most clinically consistent with very early onset inflammatory bowel disease. We further explore previous studies that implicate the underlying neuroenteric abnormalities in HSCR as possible explanations for this patient's intestinal immune and inflammatory dysregulation.
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