Treating neonatal intestinal obstruction with ostomy in continuity: a single-center comparative study

Introduction

Intestinal obstruction is the most prevalent surgical emergency in neonates. Characterized by a rapid onset and high mortality, the most common causes include congenital intestinal atresia (CIA), necrotizing enterocolitis (NEC), meconium ileus (MI), Hirschsprung’s disease (HD), intestinal malposition (IM), and anorectal malformation (1-3).

Predominant surgical procedures include intestinal resection and anastomosis, enterostomy, and abdominal drainage. Abdominal drainage is generally used as a temporary measure, with definitive surgery planned only after the patient’s condition stabilizes (4,5).

For neonates, the optimal treatment strategy for the intestine retained after diseased segment resection is primary anastomosis of the remaining intestine. This maintains intestinal continuity, preserves the function of the distal intestinal segment, and avoids a second surgery. Primary anastomosis is preferred in terms of reducing nursing difficulty, shortening the hospital stay, and reducing hospital costs (6). However, in cases where the ratio of the remaining proximal and distal intestine segment diameter is greater than 4:1 (7), or if there is severe intra-abdominal infection, direct anastomosis may lead to anastomotic leak, and the distal intestine segment may develop stenosis after treatment of NEC. In such cases, a temporary enterostomy is necessary. Enterostomy can be classified into single-barrel ostomy, double-barrel ostomy, and ostomy in continuity (OIC) (Bishop-Koop or Santulli), based on the surgical technique used.

Double-barrel or single-barrel stoma surgery has been favored in the past due to its simplicity and ease of implementation. However, intractable diarrhea frequently occurs after enterostomy among patients with a short proximal small intestine segment. This results in a significant loss of digestive juices, leading to fluid imbalance and electrolyte disorders. In such conditions, long-term total parenteral nutrition (TPN) treatment is often required, worsening the risk of sepsis, cholestasis, liver dysfunction, and venous catheter-related complications (8,9).

The OIC procedure comprises two primary techniques: the Santulli enterostomy and the Bishop-Koop enterostomy (10). Santulli’s enterostomy was initially used to treat CIA (11). In the early stages of its application, this procedure served to divert intestinal contents, relieve pressure on the anastomosis, and prevent anastomotic leaks. Once the edema at the anastomosis resolved, intestinal fluid could pass through the anastomosis into the distal intestine (12). The Bishop-Koop stoma was first used in 1955 for the treatment of meconium ileus (13). By gradually emptying the thick and dry meconium from the distal intestine segment, this technique facilitates the passage of contents from the proximal intestine segment into the distal intestine segment. This process not only improves nutrient absorption but also promotes dilation of the distal intestine segment.

Herein, the aim of this study was to assess the therapeutic effect of OIC and provide a reference for the choice of surgical approaches for intestinal obstruction. We present this article in accordance with the STROBE reporting checklist (available at https://tp.amegroups.com/article/view/10.21037/tp-24-246/rc).

Methods

Study participants

In this study, the clinical data of neonates with intestinal obstruction treated with OIC in our hospital (Anhui Provincial Children’s Hospital) between June 2019 and February 2024, were retrospectively analyzed. The study was conducted in accordance with the Declaration of Helsinki (as revised in 2013). Ethical approval for the study was obtained from the Ethics Committee of Anhui Provincial Children’s Hospital (No. EYLL-2021-010). Written informed consent was obtained from patients’ legal guardians prior to enrolment. We collected the relevant cases through the hospital’s electronic medical record system.

The criteria for inclusion of patients in this retrospective study were as follows: (I) neonates with gestational age (GA) that was corrected of less than 28 days; (II) infants who underwent ostomy in our hospital due to NEC, MI, CIA, or intestinal volvulus; and (III) infants whose subsequent closure surgery was performed in our hospital with complete follow-up records.

The exclusion criteria were as follows: (I) neonates older than 28 days of age or GA that was corrected at the time of surgery; (II) infants with other congenital diseases, such as HD, that affect the timing of subsequent stoma closure; (III) infants who did not undergo initial or subsequent surgery at our hospital; and (IV) infants lost to follow-up.

The surgical procedures in this cohort were carried out by four pediatric surgeons, all of whom possessed equivalent qualifications. Their experience and technical proficiency were closely matched. Comprehensive discussions and analyses of the pediatric patients' conditions in the perioperative period were conducted to ensure that the surgical strategies and quality were standardized and consistent. By assessing the general condition of the neonate, such as whether there is a combination of hypoproteinemia, severe infection, thrombocytopenia, the remaining healthy small intestine length, etc., the mode of operation is adopted according to the surgical guidelines/expert consensus. The neonates were divided into two groups based on their surgical procedures. We conducted follow-up with all the pediatric patients enrolled in our study via a multimodal approach, including telephone, online platforms, and traditional face-to-face outpatient clinic visits, to evaluate their growth, development, and defecation patterns.

Surgical method

The surgical approach was based on intraoperative findings. Enterostomy was used if there was a risk of anastomotic leak when the proximal and distal ends of the diseased intestine were directly anastomosed after diseased intestine segment resection.

The choice of Santulli enterostomy was made when the proximal small intestine segment was longer than 60 cm, the distal end was near the ileocecal valve, and the main purpose of the stoma was early decompression. Specifically, the proximal small intestine segment was exteriorized to create a stoma, and the distal intestine segment was anastomosed end-to-side with the proximal intestine segment about 4 cm away from the stoma. This technique was preferred for patients whose proximal diseased intestine segment was greater than 60 cm to minimize the risk of high-output stoma.

A Bishop-Koop type stoma was chosen in cases of patients whose distal intestine segment was more than 5 cm from the ileocecal valve. In this procedure, the distal small intestine segment was exteriorized to create a stoma, and the proximal intestine segment was anastomosed to the distal small intestine segment about 4 cm from the stoma.

For patients with severe intra-abdominal infection and patients in whom there was uncertainty regarding the presence or absence of additional intestinal ganglion cell dysplasia at the distal intestine segment, a double-barrel ostomy for the small intestine was performed.

A single-barrel ostomy was performed for patients whose overall health condition was poor and found unsuitable for prolonged surgery.

Observation indicators

Details pertaining to the patient’s demographics, GA, and perioperative data were collected for this study. Perioperative data included age at first surgery, weight at first surgery, weight-for-age Z-score, primary diagnosis, duration of stoma creation surgery, bleeding volume during stoma creation surgery, length of hospital stay after stoma creation surgery, duration of parenteral nutrition after stoma creation surgery, time to first defecation after surgery, postoperative complications, interval between stoma closure and stoma creation surgeries, weight at stoma closure surgery, weight-for-age Z-score at stoma closure surgery, duration of stoma closure surgery, bleeding volume at stoma closure surgery, and length of hospital stay after stoma closure surgery.

Statistical analysis

Data were analyzed using the IBM SPSS Statistics 23 (IBM Corp., Armonk, NY, USA). Chi-squared tests were used for counting data, and Fisher exact tests were used when the expected count of a cell was less than 5. Measurement data were first analyzed to determine if they followed a normal distribution, and normally distributed data were expressed as the mean and standard deviation and analyzed using the independent samples t-test. A statistical significance was defined as a P value less than 0.05 (P<0.05).

Results

There were totally 51 patients identified from our strategy. Twenty neonates underwent OIC (Santulli/Bishop-Koop enterostomy). Among the 20 in the OIC group, one neonate was transferred to another hospital at the request of the family after reoperation due to an anastomotic leak, and another was taken home by the family after the neonate was diagnosed with abnormal genetic issues. Stoma closure surgery was completed in a total of 18 patients in this group.

In the control group, 31 infants underwent enterostomies. Among them, two infants had subsequent stoma closure surgery completed at another hospital, and one child was diagnosed with HD after surgery and excluded after undergoing radical surgery for HD. A total of 28 children underwent stoma closure surgery as scheduled, comprising seven neonates who underwent single-barrel stoma surgery and 21 who underwent double-barrel stoma surgery. Therefore, the study sample consisted of 46 neonates and the flowchart in Figure 1 showed our research process.

Figure 1 The flow chart of patient inclusion in this study. OIC, ostomy in continuity; NEC, necrotizing enterocolitis.

The two groups of surgical neonates exhibited no statistically significant differences in gender, GA, age at stoma creation surgery, weight at stoma creation surgery, preoperative weight-for-age Z-score, or type of disease (Table 1).

There was no statistically significant difference between the groups with respect to the time of performing the first ostomy, intraoperative bleeding volume, length of hospital stay after stoma creation surgery, or time to the first postoperative bowel movement. This finding can be attributed to our standard protocol, which mandates a minimum interval of 12 weeks between the stoma creation and closure surgeries, alongside a minimum body weight of 5 kg (14,15).

Neonates in the OIC group had a significantly shorter duration of parenteral nutrition compared to the control group (P=0.02) (Table 2).

The two groups exhibited no statistically significant difference in terms of weight at stoma closure surgery or days of hospital stay after stoma closure surgery (Table 3). However, in comparison to the control group, neonates in the OIC group had a shorter interval between the stoma creation and closure surgeries (P=0.02), a shorter duration of surgery for stoma closure (P<0.001), less bleeding during stoma closure surgery (P<0.001). This disparity was most likely because neonates in the OIC group required only stoma closure without the need for more complex intra-abdominal procedures. And the OIC group showed better weight-for-age Z-scores than the control group (P=0.01) (Figure 2). We also noted that there was a decrease in the postoperative Z-score in the control group compared to pre-surgery Z-score.

Figure 2 Comparison of weight-for-age Z-scores before ostomy and before stoma closure surgery in the two groups. OIC, ostomy in continuity.

An analysis of ostomy-related complications revealed that in the OIC group, a total of 4 infants (20.00%) experienced stoma-related complications. Two patients (10.00%) developed high ostomy output (greater than 20 mL/kg/d) after surgery. Both cases involved neonates who underwent the Santulli procedure, with the lengths of the proximal small bowel being 70 and 50 cm, respectively, against a total small bowel length of 95 and 105 cm. After ruling out infectious etiologies for diarrhea, the neonates were initially nourished with breast milk. Upon the onset of diarrheal symptoms, their diet was transitioned to a hydrolyzed formula with reduced osmolarity, yet the symptoms of diarrhea persisted despite these interventions. The condition of one of them improved after antidiarrheal treatment and stoma blockage. In the case of the other patient, an exploratory laparotomy was performed 8 weeks after the initial stoma creation surgery due to recurrent diarrhea and minimal anal defecation, subsequent to adequate parenteral nutrition and intestinal imaging studies. One neonate (5.00%) developed both diarrhea and anastomotic obstruction. The original stoma and anastomosis site were removed intraoperatively, and a direct anastomosis of the proximal and distal intestine segments was performed. One patient (5.00%) developed postoperative adhesive intestinal obstruction, which improved after conservative treatment was initiated.

In contrast, in the control group, 16 neonates (57.14%) had stoma-related complications, 13 neonates (46.43%) developed diarrhea, and this was significantly higher than that in the OIC group. Among them 1 (3.57%) developed stoma prolapse. One infant (3.57%) was treated for surgical site infection and a stoma retraction, while 2 infants developed adhesive intestinal obstruction (7.14%) (Table 4).

Discussion

Neonatal intestinal obstruction is a life-threatening condition, most of which requires emergency treatment. Because of the time limitation or the physical condition of the child, enterostomy is an important option for which can relieve the obstruction quickly. Different from adults, enterostomy in children is mostly temporary and has many complications. Diarrhea is related to the length of the remaining normal bowel. The incidence of postoperative stoma retraction and skin infection in our center is significantly lower than that in international reports, which may be related to the surgical skills of the surgeon, the choice of incision and postoperative nursing measure (16,17).

Our study involved the use of OIC (Santulli enterostomy and Bishop-Koop enterostomy) for the treatment of neonatal intestinal obstruction and an evaluation of risks of OIC surgery. The duration of the initial surgery was slightly longer in the OIC group in comparison with the control group. This was most likely because of the more intricate surgical procedures adopted for neonates in the OIC group (18). When compared with the control group, although the OIC cohort did not exhibit superior operative time, due to the circumvention of disuse atrophy in the distal bowel segment associated with the stoma, a marked decrease in the potential for postoperative diarrhea, a reduction in the interstitial period prior to stoma closure, and the ease of care management, there is a pronounced preference for the OIC surgical approach. This preference has been corroborated through our routine communications with the caregivers of pediatric patients.

Preoperative intestinal preparation is essential before stoma closure surgery. This includes procedures such as instilling intestinal contents into the distal intestine segment in the case of patients undergoing double-barrel stoma surgery and performing anus-accessed enemas for patients prior to single-barrel stoma surgery. This has rarely been reported in the previous literature. Consequently, the interval between the stoma creation and closure surgeries in neonates of the OIC group was shorter in comparison with the control group. We did not find any significant difference between the two groups in the length of hospital stay after stoma closure surgery. This finding is inconsistent with previous reports (18). This may be due to the recovery of intestinal motility needs time.

There were no instances of cholestasis, liver function impairment, or central venous catheter-related complications. Our analysis revealed that neonates in the OIC group had a significantly lower incidence of postoperative complications in comparison with the control group, with the incidence of diarrhea being significantly lower in this group.

Nevertheless, it must be noted that in the OIC group, one infant with NEC had to undergo Santulli enterostomy during surgery, followed by an anastomotic leak on the third postoperative day. These cases were excluded from the final analysis of the study as their subsequent treatment was not administered in our hospital. In contrast, there were no such severe complications in the control group due to the absence of intra-abdominal intestinal anastomosis. This underscores the possibility that, despite fewer postoperative complications, neonates in the OIC group could still develop intestinal anastomosis-related complications. Our conclusion was that infants experiencing an intra-abdominal infection due to intestinal perforation and infants with severe intestinal inflammation and edema were at risk of developing anastomotic leaks, and hence, continuous stoma surgery was not suitable in such cases. This is consistent with the views of Yue et al. (18).

Due to the single-center design of the study, the generalizability of our findings is constrained by the small sample size. Furthermore, because of the limited number of patients who underwent Bishop-Koop enterostomy, a comparison of the advantages and disadvantages of the Santulli and Bishop-Koop enterostomies was not feasible in this study. Another limitation is that we only analyzed the nutritional status of the children through the weight Z-score and did not use the height-for-age Z-score, as some patients did not have their height measured before stoma closure. This aspect needs to be evaluated in future research. In addition, while the primary aim of this study was to offer suitable therapeutic strategies for patients presenting with severe intra-abdominal infections or compromised overall health, making them poor candidates for extended surgical procedures, and despite the fact that the selection of the control group took into account the overall health and surgical risks of the pediatric patients, the control group may still have been at a higher baseline risk for complications compared to the OIC group. We will employ appropriate statistical methodologies in future research to adjust for this disparity, thereby ensuring the precision and impartiality of the study outcomes.

Conclusions

In conclusion, our findings in this study indicated that OIC surgery was a feasible technique for addressing neonatal intestinal obstruction. As an ostomy method, OIC has a good performance in maintaining intestinal integrity, shortening parenteral nutrition time, and reducing the incidence of postoperative diarrhea. If short bowel syndrome is expected to occur during the operation, OIC is recommended. Of course, due to the existence of intraperitoneal anastomosis, the surgeon should pay attention to the occurrence of anastomotic fistula. When there are no factors affecting anastomotic healing, such as severe abdominal infection, hypoalbuminemia, and hemodynamic instability, OIC should be considered.

Acknowledgments

We would like to acknowledge the hard and dedicated work of all the staff that implemented the intervention and evaluation components of the study.

Funding: None.

Footnote

Reporting Checklist: The authors have completed the STROBE reporting checklist. Available at https://tp.amegroups.com/article/view/10.21037/tp-24-246/rc

Data Sharing Statement: Available at https://tp.amegroups.com/article/view/10.21037/tp-24-246/dss

Peer Review File: Available at https://tp.amegroups.com/article/view/10.21037/tp-24-246/prf

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://tp.amegroups.com/article/view/10.21037/tp-24-246/coif). The authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. The study was conducted in accordance with the Declaration of Helsinki (as revised in 2013), and was approved by the Ethics Committee of Anhui Provincial Children’s Hospital (No. EYLL-2021-010). Written informed consent was obtained from patients’ legal guardians prior to enrolment.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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