Development of Prediction Model for Hirschsprung-Associated Enterocolitis (HAEC) in Postoperative Hirschsprung Patients.

BACKGROUND: Despite identifying numerous risk factors for postoperative Hirschsprung-associated enterocolitis (HAEC), predicting individual risk remains challenging. This study aimed to develop a clinical prediction model for predicting the probability of postoperative HAEC within 5 years after surgery in Hirschsprung individuals. METHODS: The study included all children with Hirschsprung disease who underwent definitive surgery at Chiang Mai University Hospital from 2006 to 2021. Concomitant anorectal abnormalities and incomplete data were excluded. A multivariable logistic regression analysis, adjusted for correlated data, was utilized to develop the prediction model. RESULTS: Of the included 274 patients, 75 patients (27.4%) experienced postoperative HAEC within 5 years, totaling 121 episodes. Based on statistical and theoretical significance, eight parameters were utilized as predictors, which included male (OR1.23,95%CI:0.53-2.86), trisomy21(OR1.34,95%CI:0.21-8.45), weight at the time of surgery (OR0.86,95%CI:0.73-1.02), absence of exclusive breastfeeding (OR1.51,95%CI:0.65-3.51), length of the aganglionic segment (rectosigmoid (OR1.32,95%CI:0.48-3.62), long segment (OR41.39,95%CI:3.00-570.37), and total colonic aganglionosis (OR710.20,95%CI:23.55-21420.72)), preoperative stoma (OR1.72,95%CI:0.34-8.58), surgical approach (Duhamel (OR0.06,95%CI:0.01-0.81) and abdominal assisted trans anal endorectal pull-through (OR0.04,95%CI:0.002-0.65)), and early HAEC before two weeks following surgery (OR1.98,95%CI:0.67-5.82). The derived predictive model exhibited acceptable discriminative performance (AuROC:0.749,95%CI:0.679-0.816). Risk groups were categorized into low and high-risk, with positive likelihood ratios of 0.65 and 10.70, respectively. Recommendations for management and follow-up were generated based on these risk groups. An online application has been developed for calculating individual risk of postoperative HAEC and offering management suggestions with follow-up schedule: [https://w1.med.cmu.ac.th/surgery/personnel/pedsurgerycmu/#HAEC-Calculator]. CONCLUSIONS: This risk predictive model accurately estimates the probability of postoperative HAEC within 5 years after surgery in Hirschsprung patients. It facilitates risk stratification and provides personalized recommendations to parents for the prevention and early detection of postoperative HAEC. LEVELS OF EVIDENCE: Level II Retrospective cohort study (Prognosis study).

A comparison of the success rate of pneumatic reduction in intussusception between general anesthesia and deep sedation: a randomized controlled trial.

PURPOSE: This study was aimed to compare the success rate between patients who underwent general anesthesia and deep sedation. METHODS: Patients who were diagnosed with intussusception and had no contraindications would receive non-operative treatment first by undergoing pneumatic reduction. The patients were then split in to two groups: one group underwent general anesthesia (GA group), while the other underwent deep sedation (SD group). This study was a randomized controlled trial which compared success rate between two groups. RESULTS: A total of 49 episodes diagnosed with intussusception were random into 25 episodes in GA group and 24 episodes in SD group. There was no significant difference in baseline characteristic between the two groups. The success rates of GA group and SD group were equally 88.0% (p = 1.00). Sub-analysis of the success rate was lower in the patients with high-risk score for failed reduction. (Chiang Mai University Intussusception (CMUI) failed score in success VS failed = 6.9 ± 3.2 vs. 10.3 ± 3.0 p = 0.017). CONCLUSION: General anesthesia and deep sedation offered similar success rates. In cases of high risk of failure, general anesthesia should be considered to accommodate the switch to surgical management in the same setting if the non-operative approach fails. The appropriate treatment and sedative protocol also increase the success of reduction.

Prognostic factors of postoperative Hirschsprung-associated enterocolitis: a cohort study.

PURPOSE: To identify prognostic factors of postoperative Hirschsprung-associated enterocolitis (HAEC). METHOD: A retrospective cohort study of Hirschsprung patients between 2006 and 2021 was conducted. Patients with anorectal malformation and non-definitive surgery were excluded. Associated factors for postoperative HAEC were reported with hazard ratio (HR) and 95% confidence interval (CI). RESULTS: Forty-nine patients were excluded due to concurrent anorectal malformation and incomplete data. Of 274 patients, 75 patients (27.4%) had at least one episode of postoperative HAEC. There were 28 patients (37.3%) who had multi-episodes of HAEC. The total episodes of post-operative HAEC in this study were 121 episodes (36.8%). In multivariable survival analysis, significant factors associated with postoperative HAEC were the aganglionic level above sigmoid colon (HR = 3.47, p = 0.023, 95% CI 1.19-10.09), and total colonic aganglionosis (HR = 14.83, p = 0.004, 95% CI 2.33-94.40). The patients who experienced clinical enterocolitis before 2 weeks after surgery significantly developed more postoperative HAEC (HR = 5.32, p = 0.038, % CI 1.09-25.92). The incidence of postoperative HAEC was increase in patients with postoperative obstructive symptoms (48.0%). One patient died due to severe sepsis from postoperative HAEC, while three others required intensive care. CONCLUSIONS: The long involvement of aganglionic segment and early postoperative HAEC was significantly associated with postoperative HAEC. Frequent follow-up, parental education, and early treatment are recommended in these individuals, particularly in the first year after surgery.

Temporal Validation of Chiang Mai University Intussusception Failed Reduction Score (CMUI).

This study aimed to validate the "Chiang Mai University Intussusception Failed Score (CMUI)" for intussusception non-operative reduction. Both a 2-year retrospective and a 5-year prospective consecutive review of patients with intussusception were conducted. Data were collected from January 2013 to December 2020. Related retrospective data of a developmental set from two centers from January 2006 to December 2012 were used. Ten prespecified prognostic factors for failed reduction were collected and from these a predictive score was calculated. The actual results of non-operative reduction were collected and set as a reference standard. Altogether, 195 episodes of intussusception were found. Twenty-two patients were excluded due to contraindications; therefore, a total of 173 episodes were included in the validation dataset. The development data set comprised 170 episodes. We found that no statistical significance was found from comparing the areas under the ROC of two datasets (p-value = 0.31), while specificity of the validation set was 93.8% (88.1-97.3). This temporal validation showed a high specificity and a high affinity for prediction of failed reduction as the development dataset despite being in an era of a higher successful reduction rate. The intensive reduction protocols might be introduced among patients with high-risk scores.

Alterations of Gut Bacteria in Hirschsprung Disease and Hirschsprung-Associated Enterocolitis.

Hirschsprung-associated enterocolitis (HAEC) is a common life-threatening complication of Hirschsprung disease (HSCR). It has been proposed that gut microbiota, which have an essential role in gut-homeostasis, are associated with HAEC. Recent studies demonstrated an increase in alpha diversity of fecal microbiota over time in HSCR mice and a decrease in diversity after surgery. In addition, clinical studies have reported a reduction in bacterial richness in HSCR children after surgery. Some studies revealed a difference in microbiota between the proximal ganglionic and distal aganglionic intestine and found a difference in bacterial character between fecal and colonic specimens. HAEC studies found an increase in Proteobacteria, especially Escherichia and Enterobacteriaceae, with a decrease in Firmicutes and Bifidobacterium in HAEC patients. However, the direction of alpha diversity in HAEC patients is still controversial. The self-comparison of microbiota in treatment periods suggested that probiotics might improve gut dysbiosis and decrease the frequency of enterocolitis, but some reported contradictory findings. This review comprehensively summarizes and discusses key findings from animal and clinical data of the distinct microbiome associated with HCSR and the association of gut dysbiosis with the development of HAEC. This information should be useful in the establishment of novel interventions to improve gut dysbiosis and prevent enterocolitis in HSCR patients.

Development of Clinical Referral Score Model for Early Diagnosis of Hirschsprung's Disease in Suspected Pediatric Patients.

The diagnosis of Hirschsprung's disease (HSCR) relies on history, physical examination, and investigations. Some of investigation modalities could not be done in primary hospital. This study was aimed to develop the clinical score model for diagnosing and early referrals of HSCR, especially in areas where investigations were not available. Overall 483 consecutive suspected HSCR patients who were under 15 years old from January 2006 to December 2020 were included in this study, with 207 (42.86%) patients diagnosed with HSCR and 276 (51.14%) patients in the non-HSCR group. Five clinical parameters were included in the prediction model. The AuROC of clinical parameters, which included having an age younger than one month, male gender, the term infant, history of delayed meconium passage, and history of enterocolitis, was 72%. The prediction score ranged from 0-7, with a score 0-3 meaning a low risk to be HSCR (LHR+ = 0.37). We concluded that patients with suspected HSCR who had clinical score 4-7 had a high probability to be HSCR and, thus, it was suggested that these patients have an early referral for further investigations, which were contrast enema and rectal suction biopsy. In the case of a low probability of HSCR, clinical observation is still warranted. This clinical scoring system can be used as a screening tool to prevent delay diagnosis and complications.

A Prospective, randomized comparative study between ultrasound-guided posterior quadratus lumborum block and ultrasound-guided ilioinguinal/iliohypogastric nerve block for pediatric inguinal herniotomy.

BACKGROUND: Ilioinguinal/iliohypogastric nerve block is commonly performed to control postherniotomy pain. The posterior quadratus lumborum block has been recently described as an effective analgesic technique for pediatric low abdominal surgery. No data were found regarding the use of posterior quadratus lumborum block in comparison with the traditional ilioinguinal/iliohypogastric nerve block in pediatric inguinal surgery. AIM: This randomized assessor-blinded study compared postoperative analgesic effects between ultrasound-guided posterior quadratus lumborum block and ilioinguinal/iliohypogastric nerve block in pediatric inguinal herniotomy. METHODS: One- to seven-year-old children scheduled for unilateral open herniotomy were randomly assigned to receive either ultrasound-guided posterior quadratus lumborum block with 0.25% bupivacaine 0.5 mL/kg or ultrasound-guided ilioinguinal/iliohypogastric nerve block with 0.25% bupivacaine 0.2 mL/kg after induction of general anesthesia. The primary outcome was the proportion of patients who received postoperative oral acetaminophen. The required fentanyl in the recovery room, 24-hour acetaminophen consumption, success rate of regional blocks, block performance data, block-related complications, postoperative pain intensity, and parental satisfaction were assessed. RESULTS: This study included 40 patients after excluding four cases who were ineligible. The number of patients who required postoperative oral acetaminophen was significantly lower in the posterior quadratus lumborum block group (15.8% vs 52.6%; OR: 5.9; 95% CI: 1.3, 27.3; P = .022). The pain scores at 30 minutes, 1, 2, 6, 12, and 24 hours were similar between groups. There was no evidence of between-group differences in block performance time, the number of needle passes, block-related complications, and parental satisfaction. CONCLUSION: The posterior quadratus lumborum block with 0.25% bupivacaine 0.5 mL/kg provided better pain control than the ilioinguinal/iliohypogastric nerve block with 0.25% bupivacaine 0.2 mL/kg after open herniotomy in children. The ultrasound guidance technique for the posterior quadratus lumborum block is safe and as simple as the ultrasound-guided ilioinguinal/iliohypogastric nerve block for pediatric patients.