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Neurogastroenterology and Motility Nov 2021A significant proportion of children experience bowel dysfunction (including constipation and fecal incontinence) following surgical repair of Hirschsprung disease (HD)....
BACKGROUND
A significant proportion of children experience bowel dysfunction (including constipation and fecal incontinence) following surgical repair of Hirschsprung disease (HD). Persistent symptoms are thought to relate to underlying colonic and/or anorectal dysmotility. Manometry may be used to investigate the gastrointestinal motility patterns of this population.
PURPOSE
To (1) evaluate the colonic manometry equipment and protocols used in the assessment of the post-operative HD population and (2) summarize the available evidence regarding colonic motility patterns in children with HD following surgical repair.
DATA SOURCES
We performed a systematic review of the Cochrane Library, Embase, MEDLINE, and PubMed databases (January 1, 1980 and March 9, 2020). Data were extracted independently by two authors.
STUDY SELECTION
This systematic review was performed in accordance with Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA). Studies reporting the post-operative assessment of children with HD using colonic manometry were considered for inclusion.
RESULTS
Five studies satisfied selection criteria, providing a combined total of 496 children. Of these, 184 children with repaired HD underwent colonic manometry. Studies assessed heterogeneous populations, utilized variable manometry equipment and protocols, and reported limited baseline symptom characteristics, thus restricting comparability. All studies used low-resolution colonic manometry.
CONCLUSIONS
This systematic review highlighted the paucity of evidence informing the understanding of colonic dysmotility in the post-operative HD cohort. Current literature is limited by variable methodologies, heterogeneous cohorts, and the lack of high-resolution manometry.
Topics: Colon; Hirschsprung Disease; Humans; Manometry; Postoperative Complications
PubMed: 34214244
DOI: 10.1111/nmo.14201 -
World Journal of Gastroenterology Jul 2019A large proportion of patients with Hirschsprung disease experience persistent obstructive symptoms after corrective surgery. Persistent obstructive symptoms may result... (Meta-Analysis)
Meta-Analysis
BACKGROUND
A large proportion of patients with Hirschsprung disease experience persistent obstructive symptoms after corrective surgery. Persistent obstructive symptoms may result in faecal stasis that can develop into Hirschsprung-associated enterocolitis, a potential life-threatening condition. Important treatment to improve faecal passage is internal anal sphincter relaxation using botulinum toxin injections.
AIM
To give an overview of all empirical evidence on the effectiveness of botulinum toxin injections in patients with Hirschsprung disease.
METHODS
A systematic review and meta-analysis was done by searching PubMed, EMBASE and the Cochrane Library, using entry terms related to: (1) Hirschsprung disease; and (2) Botulinum toxin injections. 14 studies representing 278 patients met eligibility criteria. Data that were extracted were proportion of patients with improvement of obstructive symptoms or less enterocolitis after injection, proportion of patients with adverse effects and data on type botulinum toxin, mean dose, average age at first injection and patients with associated syndromes. Random-effects meta-analysis was used to aggregate effects and random-effects meta-regression was used to test for possible confounding factors.
RESULTS
Botulinum toxin injections are effective in treating obstructive symptoms in on average 66% of patients [event rate (ER) = 0.66, = 0.004, = 49.5, = 278 patients]. Type of botulinum toxin, average dose, average age at first injections and proportion of patients with associated syndromes were not predictive for this effect. Mean 7 duration of improvement after one botulinum toxin injections was 6.4 mo and patients needed on average 2.6 procedures. There was a significant higher response rate within one month after botulinum toxin injections compared to more than one month after Botulinum toxin injections (ER = 0.79, ER = 0.46, Q = 19.37, < 0.001). Botulinum toxin injections were not effective in treating enterocolitis (ER 0.58, = 0.65, = 71.0, = 52 patients). There were adverse effects in on average 17% of patients (ER = 0.17, < 0.001, = 52.1, = 187 patients), varying from temporary incontinence to mild anal pain.
CONCLUSION
Findings from this systematic review and meta-analysis indicate that botulinum toxin injections are effective in treating obstructive symptoms and that adverse effects were present, but mild and temporary.
Topics: Anal Canal; Botulinum Toxins, Type A; Constipation; Digestive System Surgical Procedures; Enterocolitis; Hirschsprung Disease; Humans; Postoperative Complications; Time Factors; Treatment Outcome
PubMed: 31333317
DOI: 10.3748/wjg.v25.i25.3268 -
The American Journal of Gastroenterology Dec 2019Although systemic sclerosis (SSc) is known to affect the gastrointestinal (GI) tract, most of the literature focuses on esophageal, small intestinal, or anorectal...
OBJECTIVES
Although systemic sclerosis (SSc) is known to affect the gastrointestinal (GI) tract, most of the literature focuses on esophageal, small intestinal, or anorectal manifestations. There have been no reviews focused on large bowel SSc complications in over 30 years. The aim of this study is to perform a systematic review of colonic manifestations and complications of SSc.
METHODS
An experienced librarian conducted a search of databases, including English and Spanish articles. The search used keywords including "systemic sclerosis," "scleroderma," and "colon." A systematic review was performed using Preferred Reporting Items for Systematic Review and Meta-Analyses guidelines. Case reports/series were screened for validity by adapting from criteria published elsewhere.
RESULTS
Of 1,890 articles, 74 met selection criteria. Fifty-nine of the 77 articles were case reports/series. The most common article topics on colonic SSc complications were constipation/dysmotility (15), colonic volvulus (8), inflammatory bowel disease (7), microscopic colitis (6), megacolon (6), and telangiectasia (6). Colonic manifestations constituted 24% of articles on GI complications of SSc. There were a total of 85 cases (84% women, with a median age of onset of colon complication of 52 years). Limited cutaneous SSc phenotype (65.6%) was more common than diffuse (26.2%). Patients frequently had poor outcomes with high mortality related to colonic complications (27%). Recent studies explore contemporary topics such as the microbiome in SSc and prucalopride for chronic constipation in SSc.
DISCUSSION
Colonic complications comprise a large proportion of the published reports on GI symptoms afflicting patients with SSc and require raised diagnostic suspicion and deliberate action to avoid potentially serious complications including death.
Topics: Colitis, Microscopic; Colonic Diseases; Constipation; Humans; Inflammatory Bowel Diseases; Intestinal Volvulus; Megacolon; Scleroderma, Diffuse; Scleroderma, Limited; Scleroderma, Systemic; Telangiectasis
PubMed: 31805016
DOI: 10.14309/ajg.0000000000000397 -
Pediatric Surgery International Apr 2022Hirschsprung's disease (HD) is a rare occurrence in premature infants, and the exact prevalence varying across studies. Thus, we conducted this study to determine the... (Meta-Analysis)
Meta-Analysis
PURPOSE
Hirschsprung's disease (HD) is a rare occurrence in premature infants, and the exact prevalence varying across studies. Thus, we conducted this study to determine the prevalence of and clinical presentation of HD in preterm infants.
METHODS
The PubMed, EmBase, and Cochrane library databases were searched for eligible studies throughout May 2021. The pooled incidence with 95% confidence interval (CI) was calculated using the random-effects model.
RESULTS
Thirty studies involved 4,557 children with HD were included. The pooled proportion of HD patients who were preterm infants was 7% (95% CI 5-8%; P < 0.001); the sensitivity analysis indicated that the pooled prevalence of preterm infants with HD ranged from 5.0% to 5.6%. Moreover, the pooled incidence of abdominal distention in HD patients was 83% (P < 0.001), that of vomiting was 61% (P < 0.001), that of bilious vomiting was 54% (P < 0.001), that of delayed meconium passage was 48% (P < 0.001), that of constipation was 44% (P < 0.001), and that of enterocolitis was 13% (P < 0.001).
CONCLUSIONS
This study reported the prevalence of HD patients who were preterm infants, and the most common presentations were abdominal distention, vomiting, bilious vomiting, delayed meconium passage, constipation, and enterocolitis.
Topics: Child; Constipation; Enterocolitis; Hirschsprung Disease; Humans; Infant; Infant, Newborn; Infant, Premature; Prevalence
PubMed: 35142873
DOI: 10.1007/s00383-021-05054-2 -
The American Journal of Tropical... Jan 2024The aims of this study were to estimate the prevalence of gastrointestinal manifestations among individuals with positive serology for Chagas disease (ChD) and to... (Meta-Analysis)
Meta-Analysis
The aims of this study were to estimate the prevalence of gastrointestinal manifestations among individuals with positive serology for Chagas disease (ChD) and to describe the clinical gastrointestinal manifestations of the disease. A systematic review with meta-analysis was conducted based on the criteria and recommendations of the Preferred Reporting Items for Systematic Reviews and Meta-Analysis guidelines. The PubMed, Scopus, Virtual Health Library, Web of Science, and Embase databases were used to search for evidence. Two reviewers independently selected eligible articles and extracted data. RStudio® software was used for the meta-analysis. For subgroup analysis, the studies were divided according to the origin of the individuals included: 1) individuals from health units were included in the health care service prevalence analysis, and 2) individuals from the general population were included in the population prevalence analysis. A total of 2,570 articles were identified, but after removal of duplicates and application of inclusion criteria, 24 articles were included and 21 were part of the meta-analysis. Most of the studies were conducted in Brazil. Radiological diagnosis was the most frequent method used to identify the gastrointestinal clinical form. The combined effect of meta-analysis studies showed a prevalence of gastrointestinal manifestations in individuals with ChD of 12% (95% CI, 8.0-17.0%). In subgroup analysis, the prevalence for studies involving health care services was 16% (95% CI, 11.0-23.0%), while the prevalence for population-based studies was 9% (95% CI, 5.0-15.0%). Megaesophagus and megacolon were the main forms of ChD presentation in the gastrointestinal form. The prevalence of gastrointestinal manifestations of ChD was 12%. Knowing the prevalence of ChD in its gastrointestinal form is an important step in planning health actions for these patients.
Topics: Humans; Gastrointestinal Tract; Chagas Disease; Brazil
PubMed: 38052078
DOI: 10.4269/ajtmh.23-0323 -
Journal of Pediatric Surgery Nov 2020To establish the cogency of recommendations for the appropriate age for pull-through and ileostomy closure in Total Colonic Aganglionosis-Hirschsprung Disease's (TCA-HD). (Meta-Analysis)
Meta-Analysis
AIM
To establish the cogency of recommendations for the appropriate age for pull-through and ileostomy closure in Total Colonic Aganglionosis-Hirschsprung Disease's (TCA-HD).
METHOD
Medline, PubMed, Cochrane, and the ClinicalKey databases were searched without date restriction. The studies that reported TCA-HD cases were evaluated for the number of cases, age at the definitive procedure, age at the ileostomy closure, reported complications, and the type of procedure. Perianal excoriation and diaper rash rates were analyzed using SPSS software, with p < 0.05 considered significant.
RESULTS
Twenty-five studies mentioned TCA-HD findings between 1968 and 2019. The total number of patients who had definitive surgery was 218. Analysis showed no correlation between development of diaper rash and the age of the patient at the time of the definitive surgery or ileostomy closure. Studies scored between six and nine of nine possible stars on the NOS scoring system.
CONCLUSION
There is no correlation between age of surgery and postoperative diaper rash. Delaying the definitive procedure or ileostomy closure for TCA-HD has limited support on a review of current studies. The perianal excoriation/diaper rash is not reported in the literature at a high enough frequency to warrant keeping a diverting ileostomy until toilet trained of urine.
TYPE OF STUDY
Systematic review and meta-analysis. Levels of evidence IV.
Topics: Anastomosis, Surgical; Hirschsprung Disease; Humans; Ileostomy; Infant; Postoperative Complications; Treatment Outcome
PubMed: 32106964
DOI: 10.1016/j.jpedsurg.2020.02.007 -
Arquivos Brasileiros de Cirurgia... 2019Many published studies have estimated the association of rs2435357 and rs1800858 polymorphisms in the proto-oncogene rearranged during transfection (RET) gene with... (Meta-Analysis)
Meta-Analysis
INTRODUCTION
Many published studies have estimated the association of rs2435357 and rs1800858 polymorphisms in the proto-oncogene rearranged during transfection (RET) gene with Hirschsprung disease (HSCR) risk. However, the results remain inconsistent and controversial.
AIM
To perform a meta-analysis get a more accurate estimation of the association of rs2435357 and rs1800858 polymorphisms in the RET proto-oncogene with HSCR risk.
METHODS
The eligible literatures were searched by PubMed, Google Scholar, EMBASE, and Chinese National Knowledge Infrastructure (CNKI) up to June 30, 2018. Summary odds ratios (ORs) and 95% confidence intervals (CIs) were used to evaluate the susceptibility to HSCR.
RESULTS
A total of 20 studies, including ten (1,136 cases 2,420 controls) for rs2435357 and ten (917 cases 1,159 controls) for rs1800858 were included. The overall results indicated that the rs2435357 (allele model: OR=0.230, 95% CI 0.178-0.298, p=0.001; homozygote model: OR=0.079, 95% CI 0.048-0.130, p=0.001; heterozygote model: OR=0.149, 95% CI 0.048-0.130, p=0.001; dominant model: OR=0.132, 95% CI 0.098-0.179, p=0.001; and recessive model: OR=0.239, 95% CI 0.161-0.353, p=0.001) and rs1800858 (allele model: OR=5.594, 95% CI 3.653-8.877, p=0.001; homozygote model: OR=8.453, 95% CI 3.783-18.890, p=0.001; dominant model: OR=3.469, 95% CI 1.881-6.396, p=0.001; and recessive model: OR=6.120, 95% CI 3.608-10.381, p=0.001) polymorphisms were associated with the increased risk of HSCR in overall.
CONCLUSIONS
The results suggest that the rs2435357 and rs1800858 polymorphisms in the RET proto-oncogene might be associated with HSCR risk.
Topics: Genetic Predisposition to Disease; Hirschsprung Disease; Humans; Polymorphism, Genetic; Proto-Oncogene Mas; Proto-Oncogene Proteins c-ret; Sensitivity and Specificity
PubMed: 31644668
DOI: 10.1590/0102-672020190001e1448 -
Fetal and Pediatric Pathology Dec 2020Previous studies have suggested a close association between REarranged during Transfection (RET) c.73 + 9277T > C and c.135G > A polymorphisms and... (Meta-Analysis)
Meta-Analysis
Association of REarranged during Transfection (RET) c.73 + 9277T > C and c.135G > a Polymorphisms with Susceptibility to Hirschsprung Disease: A Systematic Review and Meta-Analysis.
Previous studies have suggested a close association between REarranged during Transfection (RET) c.73 + 9277T > C and c.135G > A polymorphisms and Hirschsprung disease (HSCR) susceptibility. The results are inconsistent and contradictory. Thus, we performed a meta-analysis to evaluate the association of RET c.73 + 9277T > C and c.135G > A polymorphisms with risk of HSCR. The eligible literatures were searched by PubMed, Google Scholar, EMBASE, and CNKI up to August 5 2019. A total of 20 studies including 10 studies with 1136 cases and 2420 controls on c.73 + 9277T > C and 10 studies with 917 cases and 1159 controls on c.135G > A were selected. Pooled ORs revealed that c.73 + 9277T > C and c.135G > A polymorphisms were significantly associated with an increased risk of HSCR. Moreover, stratified analysis revealed that c.73 + 9277T > C and c.135G > A polymorphisms were associated with HSCR risk in Asian, Caucasian and Chinese populations. This meta-analysis result indicated that the RET c.73 + 9277T > C and c.135G > A polymorphisms were associated with susceptibility to HSCR.
Topics: Asian People; Genetic Predisposition to Disease; Hirschsprung Disease; Humans; Polymorphism, Single Nucleotide; Proto-Oncogene Proteins c-ret; Transfection
PubMed: 31590591
DOI: 10.1080/15513815.2019.1672225 -
Journal of Pediatric Gastroenterology... Apr 2021The diagnosis of Hirschsprung disease (HD) depends on the histopathological analysis of rectal biopsies. This review aims to define the best rectal biopsy technique. A... (Meta-Analysis)
Meta-Analysis
The diagnosis of Hirschsprung disease (HD) depends on the histopathological analysis of rectal biopsies. This review aims to define the best rectal biopsy technique. A systematic literature review and proportional meta-analysis of the available case series studies of rectal biopsies were performed in this study. All case series with more than five rectal biopsies in children younger than 18 years of age suspected of HD that described at least one type of rectal biopsy were included. The studies that did not specify the rate of conclusive results and the rate of complications of the biopsy procedures were excluded. According to the literature review, there were four different techniques of rectal biopsy: open, suction, punch, and endoscopic. In the title and abstract screening process, we assessed 496 articles, 159 fulfilled the eligibility criteria, and 71 studies reported our outcomes of interest and were included in the meta-analysis. The pooled proportion of conclusive results was 94% in open biopsies (95% CI 0.89-0.98), 95% in punch (95% CI 0.90-0.98), and 88% in suction group (95% CI 0.85-0.92). The pooled proportion of complication rates was 2% in open biopsies (95% CI 0.00031-0.04), 0.039% in suction (95% CI 0.00023-0.0006), and 2% in punch biopsies (95% CI 0.00075-0.04). Suction, punch, and open techniques presented comparable rates of conclusive results. In the suction group, the association between different methods of histopathological analysis increased conclusive results rates; however, the punch biopsy was associated with significantly higher complication rates than the suction technique.
Topics: Biopsy; Child; Digestive System Surgical Procedures; Hirschsprung Disease; Humans; Infant; Rectum; Suction
PubMed: 33416267
DOI: 10.1097/MPG.0000000000003041 -
Journal of Pediatric Surgery May 2024Studies examining functional outcomes and health-related quality of life for patients with congenital surgical anomalies (CSA) are increasingly common. However, the... (Meta-Analysis)
Meta-Analysis Review
PURPOSE
Studies examining functional outcomes and health-related quality of life for patients with congenital surgical anomalies (CSA) are increasingly common. However, the prevalence of mental health disorders in this population has not been determined. The purpose of this review is to summarize the reported prevalence of mental health disorders in children born with gastrointestinal CSA.
METHODS
A systematic review of the literature was conducted on Medline (OVID), EMBASE (OVID), CINAHL (EbscoHost), and Scopus to identify studies reporting mental health diagnoses in children with a gastrointestinal CSA. A meta-analysis of the prevalence of anxiety disorders and depressive disorders was completed.
RESULTS
Of 2546 manuscripts, seven met the inclusion criteria. All included manuscripts looked at patients with anorectal malformation (ARM) and/or Hirschsprung disease (HD). No studies assessed other gastrointestinal CSAs. A total of 183 psychiatric diagnoses were reported in 1167 patients. A pooled analysis of ARM patients revealed a 14.0% prevalence of anxiety disorders (N = 208; CI = 4.4-23.4%) and an 18.75% prevalence of depressive disorders (N = 48; CI = 7.7-29.8%). A pooled analysis of HD patients revealed a 15.8% prevalence of anxiety disorders (N = 19; CI = 0.6-32.2%) and a 4.75% prevalence of depressive disorders (N = 758; CI = 3.23-6.26%).
CONCLUSIONS
There is a paucity of evidence on mental health outcomes of individuals born with gastrointestinal CSA. However, the available evidence suggested a high prevalence of mental health diagnoses in individuals born with ARM or HD with 1 in 7 having an anxiety disorder. Additionally, 1 in 5 ARM patients and 1 in 20 HD patients were diagnosed with a depressive disorder.
TYPE OF STUDY
Systematic review and meta-analysis.
LEVEL OF EVIDENCE
IV.
Topics: Child; Female; Humans; Mental Health; Prevalence; Quality of Life; Mental Disorders; Anorectal Malformations; Hirschsprung Disease; Anxiety
PubMed: 38388285
DOI: 10.1016/j.jpedsurg.2024.01.047