Prevention and management of recurrent postoperative Hirschsprung's disease obstructive symptoms and enterocolitis: Systematic review and meta-analysis

Soh, Han Jie; Nataraja, Ramesh M.; Pacilli, Maurizio

doi:10.1016/j.jpedsurg.2018.08.024

Cited by 42 publications

(37 citation statements)

References 38 publications

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“…Our results indicated that duration of improvement of obstructive symptoms was on average six months and that most patients need on average two to three injections. This in line with previous meta-analytic findings and with evidence on the effectiveness of BT injections in other patient groups, including chronic constipation, anal fissures and internal sphincter achalasia[4,6,23]. In addition, evidence from three studies (all included in our meta-analysis), indicated that short term response was predictive for long-term response, although studies used different cut-off points for defining short- and long term response[7-9].…”

Section: Discussionsupporting

confidence: 91%

“…A meta-analysis on different treatment strategies for obstructive symptoms showed short-term improvement after BT injections in 77% of patients and decreased to 43% of patients in the long-term[6]. However, that meta-analysis did neither draw conclusions on effects on the prevalence of enterocolitis, nor on the complication rate and adverse events after BT injections.…”

Section: Introductionmentioning

confidence: 99%

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Botulinum toxin injections after surgery for Hirschsprung disease: Systematic review and meta-analysis

Roorda¹,

Abeln²,

Oosterlaan³

et al. 2019

WJG

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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, P = 0.004, I 2 = 49.5, n = 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, vs ER = 0.46, Q = 19.37, P < 0.001). Botulinum toxin injections were not effective in treating enterocolitis (ER 0.58, P = 0.65, I 2 = 71.0, n = 52 patients). There were adverse effects in on average 17% of patients (ER = 0.17, P < 0.001, I 2 = 52.1, n = 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.

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Section: Discussionsupporting

confidence: 91%

Section: Introductionmentioning

confidence: 99%

Botulinum toxin injections after surgery for Hirschsprung disease: Systematic review and meta-analysis

Roorda¹,

Abeln²,

Oosterlaan³

et al. 2019

WJG

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“…Similarly, at our institution, we take a measured approach to botulinum toxin IAS injection and reserve it for patients with an anastomosis without evidence of stricture and ongoing obstructive type symptoms postoperatively. A recent meta-analysis of 2 case-control studies, Soh et al 69 reported no difference in the incidence of postoperative stricture with routine dilations. Additionally, they reported decreased incidence of enterocolitis with routine postoperative rectal irrigations implemented for a duration of 1 to 2 weeks or 10 days.…”

Section: Postoperative Management and Expectationsmentioning

confidence: 98%

“…Additionally, they reported decreased incidence of enterocolitis with routine postoperative rectal irrigations implemented for a duration of 1 to 2 weeks or 10 days. 69 However, controlled prospective studies are lacking. In our center, we do not implement routine irrigations postoperatively; however, families are taught the irrigation technique prior to discharge.…”

Section: Postoperative Management and Expectationsmentioning

confidence: 99%

Surgery, Surgical Pathology, and Postoperative Management of Patients With Hirschsprung Disease

2019

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Endorectal pullthrough surgery is integral in the treatment of patients with Hirschsprung disease. Several different surgical procedures exist, which share as common goals to excise the aganglionic segment and upstream transition zone and attach ganglionic bowel just proximal to the anal canal. The operation requires collaboration between surgeon and pathologist to localize ganglionic bowel and prevent retention of transition zone. Intraoperative frozen sections are extremely important, first to establish that ganglion cells are present and subsequently to exclude features of transition zone (partial circumferential aganglionosis, myenteric hypoganglionosis, and submucosal nerve hypertrophy) at the proximal surgical (anastomotic) margin. Postoperative histopathological analysis of resection specimens should be tailored to document distal aganglionosis, document the length of the aganglionic segment and its proximity to the anastomotic margin, and confirm that transition zone has been resected completely. Adherence to the recommendations described in this review will reduce the likelihood of transition zone pullthrough and should decrease the incidence of persistent postoperative obstructive symptoms.

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“…As a result of abnormal intestinal motility that allows bacterial stasis and microflora dis-equilibrium, coupled with an innate immune response and several other mechanisms from the level of gene expression to the biochemical imbalance of the digestive tract, will cause intestinal barrier complex disorders that will appear as histopathological changes in bowel wall structure. 4,5,[7][8][9][10][11] Other theories that have been proven are the retention of mucin in the colonic mucosa, as well as the shift from acidity of the mucin to neutral. 12 It was proposed that HAEC patients will sustain significant changes in theirbowel tissue characterized by mucin retention and dilatation of crypts which becomes the basis of HAEC histopathological classification.…”

Section: Introductionmentioning

confidence: 99%

Histopathological changes and Hirschsprung’s associated enterocolitis (HAEC) scores

et al. 2020

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Histopathological changes that are usually permanent in the bowel of Hirschsprung's disease (HRSC) in children make susceptibility to the incidence of enterocolitis even after definitive therapy. The pathophysiology of HRSC is characterized by specific signs, and symptoms. Scoring system has been applied in diagnosis of Hirschsprung's associated enterocolitis (HAEC) due to its simplest and easiest. However, after a decade it has been several evaluated to obtain optimal benefits in the clinical setting. The aim of this study was to investigate the relationship between histopathological grade of enterocolitis and HAEC scores. Children with HRSC who underwent leveling colostomy or pull-through were sampled from the ganglionic segment. Those with a histopathological description of HAEC were included in the study. HAEC scores were evaluated retrospectively through medical records and interviews. Data were analyzed using the Spearman's Rank test. Twenty eight samples were involved in this study. The population was dominated by male (82.1%) and almost half the population (42.9%) were infants. The distribution of the histopathological grade was fairly even, except for grade V where it was only 7.2% of the total sample. Grade I & III samples were 21.4%, while grade II & IV were 25%, respectively. A significant relationship between histopathological grade and HAEC score was obtained (r = 0.927; p<0.0001). In conclusion, there is a relationship between the histopathological grade of enterocolitis and HAEC scores.

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Prevention and management of recurrent postoperative Hirschsprung's disease obstructive symptoms and enterocolitis: Systematic review and meta-analysis

Cited by 42 publications

References 38 publications

Botulinum toxin injections after surgery for Hirschsprung disease: Systematic review and meta-analysis

Botulinum toxin injections after surgery for Hirschsprung disease: Systematic review and meta-analysis

Surgery, Surgical Pathology, and Postoperative Management of Patients With Hirschsprung Disease

Histopathological changes and Hirschsprung’s associated enterocolitis (HAEC) scores

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