Abstract
Background:
Laparoscopy has been widely used in treatment of pediatric intussusception. However, controversy still exists on the best surgical approach for children with multiple recurrences. This cohort study is aimed to compare the efficacy and safety of laparoscopic ileopexy versus laparoscopic simple reduction for treatment of ileocolic intussusception with three or more episodes of recurrence in pediatric patients.
Patients and Methods:
This study enrolled 74 children with three or more recurrences of ileocolic intussusception treated at our department between January 2016 and June 2018. Of these children, 37 cases undergoing laparoscopic ileopexy were matched with 37 cases undergoing laparoscopic simple reduction according to age. Data on patient characteristics, duration of follow-up, operative parameters, and recurrence rate after laparoscopic operation were reviewed and analyzed.
Results:
Patient characteristics, including age, gender, weight, number of recurrences at surgery, clinical manifestations, and duration of follow-up, were well comparable between the two groups (each P > .05). During follow-up, recurrence occurred in 16 (43.24%) children undergoing laparoscopic simple reduction, whereas recurrence occurred in 1 (2.70%) case undergoing laparoscopic ileopexy. In comparison, laparoscopic ileopexy significantly decreased the risk of recurrence (P < .001). No statistical difference was observed between the two groups in terms of operative time, length of postoperative stay, and intra- and postoperative complications (each P > .05). No morbidity was documented in both groups.
Conclusions:
Laparoscopic ileopexy is effective and safe as a surgical approach for multiple recurrences of ileocolic intussusception in pediatric patients. The underlying mechanisms are yet to be fully clarified.
Introduction
Intussusception is defined as a segment of bowel (the intussusceptum) invaginates into the lumen of another segment of bowel. 1 It can occur in any part of the bowel, but ileocolic intussusception, which arises at the junction between the ileum and the cecum, is the most common type in the pediatric population. Intussusception is one of the leading causes of bowel obstruction in infants and young children. With delayed diagnosis and treatment, the involved part of bowel, with decreased blood supply, can progress to perforate, resulting in passage of its contents into the abdominal cavity, or other severe complications. 2 The main management of pediatric intussusception includes contrast enema and surgical intervention. Contrast enema, including air, barium, or saline enema, has been widely preferred as initial management of intussusception. 3 Surgical intervention is generally indicated when appropriate radiological facilities are unavailable; or when contrast enema fails; or when children present symptoms of perforation, shock, or peritonitis; or when a pathologic lead point (PLP) is suspected preoperatively.2,4
Recurrent intussusception is referred to intussusception occurring after a successful reduction. 5 According to a recent meta-analysis, the overall rate of recurrence is up to 12.7% after contrast enema. 3 Of note, the risk of recurrence appears to increase with each subsequent episode, and is much higher in children with three or more recurrences than those with one or two episodes. 6 Some publications have reported several potential risk factors for the recurrence of intussusception, whereas contrary findings still exist.7–9 Although PLPs are considered to be associated with recurrence in a few studies,6,9 the majority of pediatric cases with multiple recurrences do not have an identifiable etiology.2,10 The character of idiopathic etiology leads to great difficulty in exactly predicting the pattern of recurrence and identifying children who are at an increased risk of recurrence. The possibility of recurrence always increases parents' psychological stress and anxiety.11,12 Besides, multiple recurrences could increase radiation exposure resulted from repeated contrast enemas, and bring negative impact to psychological and physical development of children. 13
Management of multiple recurrences remains challenging to pediatric surgeons. Although surgical intervention has been recommended for recurrent intussusception, controversy still remains. 6 Some experts argue that prophylactic operation for multiple recurrences may be involved with overtreatment, as repeated nonsurgical reduction is effective and safe. 3 In their opinion, each recurrence could be treated as if it was the first episode.3,7,14 On the contrary, some experts support that surgical intervention is more appropriate for recurrent intussusception than nonsurgical enema, with advantages of allowing detecting PLPs, and lowering the risk of subsequent recurrence.10,15 However, few studies have evaluated the application of surgical intervention particularly in children with multiple recurrences.
Fixation of the terminal ileum to the cecum or peritoneum after manual reduction, also called ileopexy, has been proposed to be an optional surgical strategy for preventing recurrence of intussusception.11,16–19 In 2009, Chang et al. 11 reported their experience on laparoscopic fixation of the terminal ileum to peritoneum in 6 children with recurrent intussusception, and no subsequent recurrence was observed during postoperative follow-up. Nevertheless, ileopexy is not a routine surgical maneuver at present, and convincing data are still deficient regarding its efficacy and safety in children with multiple recurrences of intussusception. Herein, we conduct a retrospective cohort study directly comparing laparoscopic ileopexy versus conditional laparoscopic reduction to address this issue.
Patients and Methods
Study population
Medical records of all children treated with intussusception in our center between January 2016 and June 2018 were retrospectively reviewed. During this period, intussusception was diagnosed primarily by ultrasonography. For children with three or more recurrences, the choice of air enema or laparoscopic reduction as initial treatment was determined by patients or guardians with full informed consent. However, emergent surgical intervention was necessitated when contrast enema failed; or when children presented symptoms of perforation, shock, or peritonitis; or when children were admitted >48 hours after the onset of clinical symptoms.
In this study, children were eligible for enrollment if they satisfied the following criteria: (1) undergoing laparoscopic ileopexy or laparoscopic simple reduction, (2) having three or more recurrences at surgery, and (3) anatomical diagnosis of ileocolic intussusception confirmed by intraoperative findings. Exclusion criteria were (1) having received surgical intervention or having history of previous abdominal surgery, (2) presence of a PLP or other comorbidities confirmed by intraoperative findings, (3) peritonitis, which is one of the contraindications for ileopexy, and (4) undergoing bowel resection and anastomosis. Finally, a series of 37 children undergoing laparoscopic ileopexy were identified as the ileopexy group, whereas an equal number of children (n = 37) undergoing laparoscopic simple reduction were enrolled as the control group. Matching criterion was age at surgery (<1 year: birth date ±1 month; 1–2 years: birth date ±3 months; >2 years: birth date ±6 months), to balance potential bias. When more than one subject meets the matching criteria, controls were chosen at random.
After discharge, all children were followed up at the clinic or by telephone. When recurrence was diagnosed by radiological examinations, children were admitted immediately.
Surgical technique
Before surgery, written consent was obtained from patients or legal guardians of each child. All laparoscopic operations, either ileopexy or simple reduction, were done by an experienced surgeon team in our center. In principle, the choice of laparoscopic ileopexy or simple reduction was determined on individual preference and intraoperative findings without specific criteria, but ileopexy was not performed in the case with peritonitis, or bowel resection.
The patient was placed on the operating table in a supine position; the display was placed on the right of the operating table. A 5-mm port for laparoscope was inserted through an umbilical skin incision, and carbon dioxide pneumoperitoneum with a pressure of 6–10 mmHg was established. First, the cecum was identified and the small bowel was carefully examined for evidence of intussusception. Second, intussusception reduction was accomplished through taxis and traction with traumatic bowel graspers. In cases of tight incarceration, intraoperative air reduction was done by inserting one Foley tube into the rectal vault and by slowly pushing in an adequate amount of air, and then pulling the intussusceptum out gently. Third, the abdominal cavity was inspected carefully under laparoscopic visualization for bowel necrosis, PLPs, or other comorbidities. Then, appendectomy was performed routinely. In the ileopexy group, the cecum, the base of appendix, and the terminal ileum were sutured to the lateral peritoneum (ileopexy) with 4-0 nonabsorbable sutures, as described previously. 20
Data collection
Data of all children enrolled were retrospectively collected, including age, gender, weight, date of surgery, number of recurrences before surgical intervention, clinical manifestations, length of follow-up, operative type (laparoscopic simple reduction, or laparoscopic ileopexy), operative time, length of postoperative stay, intra- and postoperative complications, mortality, and surgical recurrence.
Statistical analysis
All statistical analyses were performed using STATA version 12.0 software. Categorical variables were expressed as frequencies and proportions (%). Continuous variables are expressed as mean ± standard deviations. The distributions were tested with the Shapiro–Wilk statistic. For intergroup comparison, Student's t-test or Mann–Whitney rank sum test was used for continuous variables as appropriate, whereas the Fisher's exact test was used for categorical variables. A two-tailed P < .05 indicated statistical significance.
Results
Patient characteristics
Table 1 summarizes the baseline characteristics of all cases in the two groups. There were no statistical differences in terms of age (P = .901), number of previous recurrences (P = .309), gender (P = .794), and weight (P = .558), as well as clinical manifestations (all P > .05), respectively. The mean follow-up period was 21.84 ± 7.51 months in the ileopexy group, and 23.30 ± 7.16 months in the control group. No statistical significance was observed (P = .395).
Patient Characteristics
Operative parameters and clinical outcome
As displayed in Table 2, the operative time was 55.05 ± 14.07 minutes in the ileopexy group and 49.19 ± 16.47 minutes in the simple reduction group. In comparison, laparoscopic ileopexy slightly prolonged operative time, but no statistical difference was reached (P = .104). Furthermore, length of postoperative stay was comparable between the two groups (P = .768). There were no statistical differences in terms of intra- and postoperative complications. No mortality was documented in both groups.
Operative Parameters and Clinical Outcome
Of note, subsequent recurrence occurred in 16 (43.24%) cases undergoing simple reduction, whereas recurrence occurred in 1 (2.70%) case undergoing ileopexy. Compared with simple reduction, laparoscopic ileopexy significantly reduced the subsequent recurrence risk (P < .001).
Discussion
Intussusception in children is a common medical emergency that requires prompt diagnosis and management. After successful nonsurgical or surgical reduction, the risk of recurrence still threatens.5,6 According to the literature, the possibility of a subsequent recurrence after the forth episode ranges from 50% to 100%.6,21 In this study, we analyzed the efficacy and safety of laparoscopic ileopexy versus laparoscopic simple reduction in children with three or more recurrences. Patient characteristics, including several possible risk factors of recurrence, were well balanced between the two groups. Compared with laparoscopic simple reduction, laparoscopic ileopexy significantly lowered the risk of recurrence, without postponing postoperative recovery or causing more surgical complications.
Despite widespread agreement on contrast enema as initial management of intussusception, instead of surgical intervention, debate persists on the appropriate treatment for multiple recurrences.14,16,21 With noting a rather high rate of subsequent recurrence, some experts have recommended surgical intervention after the third episode of intussusception.11,21 Compared with contrast enema, earlier studies reported that the rate of recurrence was lower after open reduction, and was much lower after resection. 17 In cases with multiple recurrences, the surgeon may be concerned about whether a PLP is present or whether significant injury to the intestine could be caused. 6 In addition to the removal of PLPs, local adhesion after surgical intervention was also thought to be associated with lower recurrence rate. 22 Compared with contrast enema, there are several common disadvantages of traditional laparotomy, such as surgical trauma, surgical scar, and prolonged recovery. These disadvantages hammer the application of surgical intervention for preventing recurrence. 17 Therefore, in most occasions, nonsurgical approaches are still preferred for recurrent intussusception.
The advancement of laparoscopic equipment relieves opposing attitudes toward prophylactic operation for recurrence of intussusception to some degree. As well known, laparoscopy could minimize surgical trauma, reduce postoperative pain, promote recovery, and improve cosmesis when compared with laparotomy.10,23 In children with definite operative indications, a few studies have demonstrated that children receiving laparoscopic reduction have faster oral feed, shorter hospitalization, and less postoperative complications than those receiving open reduction,4,10,15 which suggests that laparoscopy appears to be superior to laparotomy as surgical approach for intussusception.
Although the recurrence rate is lower after surgical reduction than contrast enema, it is not totally eliminated. Besides, several studies reported the recurrent rate was slightly higher after laparoscopic reduction than laparotomy,22,23 although other studies reported comparable recurrence rates between laparoscopic and open reduction. 24 A possible explanation may be that less adhesion after laparoscopic operation than laparotomy could lessen the preventive effect of surgical intervention.
Ileopexy, referring to fixation of the terminal ileum to the peritoneum or cecum, is designed to fix bowel loops in different ways, and has been used as a procedure to prevent recurrence.11,16 The underlying logic is that ileopexy limits abnormal peristalsis and theoretically prevents the mobility necessary for intussusception.16,18 In 2006, Koh et al. 17 reported their experience on fixation of the terminal ileum to ascending colon in a parallel manner, and found that this technique was not better than simple manual reduction in preventing recurrence. Using the same maneuver of fixation, another surgeon team reported that the recurrence rate was lower in children undergoing ileopexy than those undergoing simple manual reduction, but the difference did not reach statistical significance. 16 They inferred that this technique possibly hindered terminal ileum from pushing into the colon, but increased the feasibility of proximal segment pushing into the fixed terminal ileum, which thus counteracted the preventive effect on recurrence.
As aforementioned, Chang et al. 11 reported that they performed ileocolic fixation to lateral peritoneum after laparoscopic reduction in 6 children with multiple recurrences, and no subsequent recurrence occurred during follow-up. Nevertheless, small sample size and the absence of controls in their study limited definitive conclusions. In the past several years, we have attempted to apply this technique in laparoscopic operation for children with three or more recurrences. This study provides evidence that ileocolic fixation is more effective than simple reduction in reducing the risk of subsequent recurrence after three or more recurrences. In addition, this procedure did not prolong postoperative recovery or cause more surgical complications than simple reduction, which further proved its safety. Given that less adhesion could limit the preventive effect of laparoscopic operation, 22 we speculate that ileopexy possibly compensates this aspect. However, more studies with larger sample sizes are still required to confirm our conclusions.
Some experts concern that surgical intervention is involved with overtreatment. In our opinion, it is vital to determine optimal cases for laparoscopic ileopexy. First, in our institution, ileopexy is considered only for children with three or more recurrences, which is corresponding with the popular view regarding appropriate surgical timing of recurrent intussusception. 21 Second, the final choice of nonsurgical or surgical treatment is dependent on children's parents or guardians with full consent. Although parents are predisposed to choose surgical management after multiple recurrences, in fact, we also have encountered many parents who insist on nonsurgical enema and reject surgery in our center. Furthermore, it needs to be emphasized that peritonitis should be considered as one contraindication of ileopexy, to avoid possible surgical complications, such as serosal tearing, the spread of infection, adhesive bowel obstruction, and surgical recurrence attributed to lose knots after regression of bowel edema. 20 Of note, peritonitis, or other complicated situation, appears less at multiple episodes than the first or second episode, which may be associated with patients' awareness and quick consult. 12
As for inconsistent findings in studies comparing ileopexy versus simple reduction,11,16–18 we infer that several points could explain this issue. First, different procedures of ileopexy were performed in these studies, and could influence bowel peristalsis in different ways, which thus produced heterogeneous effect on subsequent recurrence. Second, in the majority of previous studies,16–18 surgical intervention was given mostly in cases with irreducible intussusception, or complicated situation, such as peritonitis, instead of cases presenting with repeated recurrence. On one hand, bowel edema might tend to be more severe in cases undergoing ileopexy in these studies, and thus increased the possibility of postoperative recurrence. In contrast, the recurrent rate significantly differed after each episode, and was relatively lower at first recurrence than multiple recurrences, 6 which possibly concealed the statistical significance. However, few studies have directly compared the efficacy between different procedures of ileopexy, and further investigation is required.
There are several limitations in our studies. First, it is a single-center study, and more studies are required to confirm our findings. Second, we have not directly compared between different procedures of ileopexy in cases with multiple recurrences, because the procedure of fixation to ascending colon, as reported in several previous studies,16–18 was performed in a rather small number of children in our center, which hindered valuable analysis. Third, longer follow-up is yet needed. Despite these limitations, our study could provide vital experience for surgeons in treating multiple recurrences and may help guide decision for management of recurrent intussusception.
Conclusions
In summary, this study suggests that children with three or more recurrences of intussusception have a significantly lower recurrent rate after laparoscopic ileopexy than simple reduction, with no statistical differences observed in terms of operating time, time to oral diet, and length of stay, as well as intra- and postoperative surgical complications. Laparoscopic ileopexy is an effective and safe approach, and may be more suitable for multiple recurrences than simple reduction.
Footnotes
Disclosure Statement
No competing financial interests exist.
Funding Information
This study was sponsored by the key research and development plan (Social Development) of Huai'an City (HAS201615) and the Maternal and Child Health Research Project of Jiangsu Province (F201713).
