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Loop versus divided colostomy for the management of anorectal malformations
Journal of Pediatric Surgery 49 (2014) 87–90
Contents lists available at ScienceDirect
Journal of Pediatric Surgery journal homepage: www.elsevier.com/locate/jpedsurg
Loop versus divided colostomy for the management of anorectal malformations Omar Oda, Dafydd Davies, Kimberly Colapinto, J. Ted Gerstle ⁎ Division of General and Thoracic Surgery, The Hospital for Sick Children, Toronto, ON, Canada
a r t i c l e
i n f o
Article history: Received 17 September 2013 Accepted 30 September 2013 Key words: Anorectal malformation Loop colostomy Divided colostomy
a b s t r a c t Purpose: The purpose of this study was to compare the clinical outcomes of loop and divided colostomies in patients with anorectal malformations (ARM). Methods: We performed a retrospective cohort study reviewing the medical records of all patients with ARM managed with diverting colostomies between 2000 and 2010 at our institution. Independent variables and outcomes of stoma complications were analyzed by parametric measures and logistic regression. Results: One hundred forty-four patients managed with a colostomy for ARM were evaluated (37.5% females, 50.7% loop, 49.3% divided). The incidence of patients with loop and divided colostomies who developed stoma-related complications was 31.5 and 15.5%, respectively (p=0.031). The incidence of prolapse was 17.8 and 2.8%, respectively (p=0.005). Multivariable-logistic regression controlling for other significant independent variables found loop colostomies to be positively associated with the development of a stoma complication (OR 3.13, 95%CI (1.09, 8.96), p=0.033). When individual complications were evaluated, it was only stoma prolapse that was more likely in patients with loop colostomies (OR 8.75, 95%CI (1.74, 44.16), p=0.009). Conclusion: Because of the higher incidence of prolapse, loop colostomies were found to be associated with a higher total incidence of complications than divided stomas. The development of other complications, including urinary tract infections (UTIs) and megarectum, were independent of the type of colostomy performed. © 2014 Elsevier Inc. All rights reserved.
Since the first diverting stoma performed for the treatment of imperforate anus in 1783 [1], the site and the type of the least troublesome stoma in the surgical management of anorectal malformations (ARM) have been major subjects for discussion amongst pediatric surgeons. Clinical studies have established the high morbidity associated with neonatal colostomy and that transverse colostomy has a higher rate of complications than sigmoid colostomy [2,3]. However debate continues regarding the type of the diverting colostomy. This study compares clinical outcomes of loop and divided colostomies performed as part of the surgical management of ARM. 1. Materials and methods Research Ethics Board approval (Application No: 1000031264, Protocol No: 1000020656) was obtained to perform a retrospective cohort study to review the medical records of all patients who presented with ARM and were managed with a diverting colostomy between November 2000 and November 2010. Patients with ARM who underwent primary corrective surgery without a diverting colostomy were not included in this study. Data were collected from ⁎ Corresponding author. Division of General and Thoracic Surgery, The Hospital for Sick Children, 555 University Avenue, Room 1526, Toronto, Ontario M5G 1X8, Canada. Tel.: +1 416 813 7500; fax: +1 416 813 7477. E-mail address: [email protected] (J.T. Gerstle). 0022-3468/$ – see front matter © 2014 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jpedsurg.2013.09.032
admission, progress, operative, discharge, and follow up clinic notes. Radiology reports were also reviewed. Demographic data collected included sex, type of ARM, and age at stoma creation. From the operative reports we determined whether a loop or divided colostomy was created. A loop colostomy was defined when the bowel wall continuity was partially preserved. A divided colostomy was defined when bowel continuity was completely disrupted regardless of the distance between the proximal and the distal ends of the stoma. For statistical purpose, stoma creation was considered early when age at stoma creation was less than 6 days, and late when age at stoma creation was equal or more than 6 days. The level of the colostomy (sigmoid, descending or transverse colon), the start of stoma function (days), and the period of time (months) the patient had the stoma were also determined. For those patients who had repair of the ARM at our institution, we noted whether a distal contrast study was performed prior to anorectoplasty and whether it showed the presence of a megarectum, as determined from the radiologist’s report. We determined if the patients developed complications from their stomas (for the time from stoma creation till stoma closure); these complications included retraction, prolapse, parastomal hernia, obstruction, and need for revision. The development of megarectum and UTIs were particular variables of interest; one of our goals was to establish if there was a relationship between them and the type of the colostomy, hence they were studied as potential stoma complications. For those who had repair of the ARM at
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our institution we documented if these patients developed any complications following anorectoplasty other than stoma complications. These included wound infection, anal stricture that required dilatation under general anesthesia or repeated anoplasty, and recurrence of recto-urinary or recto-genital fistula. A more detailed review of patients who developed UTIs was also performed; data included the presence of associated urinary tract malformations or dysfunction and wether these UTI episodes occurred before or after anorectoplasty and division of recto-urinary fistula. For those who had their stomas closed at our institution, we documented if they developed wound infections after closure of the colostomy. Statistical analysis was performed using STATA SE™ Version 10.0. The demographics and rates of complications were compared between patients initially managed with loop stomas to those managed with divided stomas. Categorical and continuous variables were analyzed with Fisher’s Exact and Wilcoxon Rank Sum Tests, respectively looking for differences between the two groups. Logistic regression analysis was performed. Along with the type of colostomy (loop versus divided), independent variables of sex, type of ARM, age at stoma creation, level of stoma, and duration of stoma were evaluated for associations with the outcomes of stoma complications (retraction, prolapse, parastomal hernia, obstruction, need for stoma revision, and the development of megarectum and UTIs). We also examined their impact on complications following ARM repair and stoma closure including wound infection, development of anal stricture or recurrent fistula. In order to prevent the inclusion of variables with no outcome association in our multiple variable logistic regression analysis, only independent variables with associations meeting significance levels of p less that 0.1 were included to control for their effects. This was repeated for each outcome measure if the number of events was sufficient [4]. The results of the multiple variable logistic regression analysis were only considered significant if they reached a p-value less than 0.05. 2. Results Over the 10 year study period 461 children with all types of ARM (low, intermediate, and high) were treated at our institution. 317 patients underwent a primary repair of the ARM without a colostomy and were excluded from the study. 144 patients were managed with a colostomy. 73 (50.7%) patients had a loop colostomy, and 71 (49.3%) patients had a divided colostomy. 112 (77.7%) had sigmoid colostomy, 14 (9.7%) had descending colostomy, and 18 (12.5%) had transverse colostomy. 138 (95.8%) patients underwent anorectoplasty at our institution: 112 underwent a posterior saggital anorectoplasty (PSARP), 18 underwent a laparascopic assisted anorectoplasty, and in 7 patients anorectoplasty was performed with an open abdominoperineal approach. The operative report was not found for one patient and we were unable to determine what type of anorectoplasty he had, despite it being performed at our institution. These, and the demographic data are shown in Table 1. 118 (85.5%) of repaired patients underwent a distal contrast study prior to anorectoplasty, and 130 (94.2%) had their colostomies closed at our institution. With respect to demographics and other data shown in table 1 including age at stoma creation (early vs. delayed), there were no statistically significant differences between patients who had a loop and those who had a divided colostomy. Stoma related complications occurred in 34 (23.6%) patients (some with more than one) giving a total of 61 complications in 144 patients. The types and rates of complications are summarized in Table 2. Patients with loop stomas were significantly more likely to develop complications (p=0.031) and the number of complications was higher in this group (0.002). When comparing the rates of developing each individual complication, only the rate of stoma prolapse was found to be statistically higher in patients with loop stomas (p=0.005).
Table 1 Demographics. Variable
Loop
n (%) Sex Male, n (%) Type of Anorectal Malformation Perineal Fistula, n (%) Vestibular Fistula, n (%) Cloaca, n (%) Recto-urethral Fistula, n (%) Recto-Bladder Neck Fistula, n (%) H-Type Fistula, n (%) Atresia without Fistula, n (%) Level of Colostomy Sigmoid, n (%) Descending, n (%) Transverse, n (%) Age at Colostomy, median (IQR) (days) Delayed Stoma Creation N 6 days old, n (%) Duration of Stoma, months (n=130), median (IQR) Type of Anorectoplasty (n=138) PSARP, n (%) Laparoscopic Assisted anorectoplasty, n (%) Abdominoperineal Approach, n (%) Undefined
73 (50.7) 71 (49.3) 144 (100%) 42 (46.7) 48 (53.3) 90 (62.5) 0.232 0.600 4 (5.5) 8 (11.3) 12 (8.3) 16 (22.0) 11 (15.5) 27 (18.8) 7 (9.6) 8 (11.3) 15 (10.4) 26 (35.6) 22 (31.0) 48 (33.3) 6 (8.2) 7 (9.9) 13 (9) 2 (2.7) 0 (0) 2 (1.4) 12 (16.4) 15 (21.1) 27 (18.8) 0.615 56 (76.7) 56 (78.9) 112 (77.8) 6 (8.2) 8 (11.3) 14 (9.7) 11 (15.1) 7 (9.9) 18 (12.5) 2 (1, 3) 2 (1, 3) 2 (1, 2) 0.914
Divided
Total
p-value C C
C
⩖
17 (22.3) 13 (18.3) 30 (20.8)
0.540 C
8 (6, 10)
0.085 ⩖
7 (5, 10)
8 (6, 10)
0.091 C 61 (87.1) 51 (75.0) 112 5 (7.1) 13 (19.1) 18 3 (4.3) 1 (1.4)
4 (5.9) 0 (0)
7 1
C = Fisher’s Exact test. ⩖ = Wilcoxon Rank Sum Test.
Anorectoplasty was performed at our institution on 138 patients. There were a total of 26 complications following anorectoplasty which occurred in 22 (15.3%) patients. These are summarized in Table 3 and were no more frequent in either group. The results of the univariable logistic regression found that sex, type of ARM, age at stoma creation, and duration of the stoma were neither positively associated with the development of complications nor the development of any of the specific complications evaluated; having not met our predetermined level of significance (p=0.1) they were not included in the multivariable analysis. The level of colostomy was found to be positively associated with the development of stoma complications. As shown in Table 4, sigmoid colostomies were most favorable, whereas transverse colostomies had the highest association with the development of complications (p=0.006, OR 4.33, 95% CI (1.53, 12.24)). Again, prolapse accounted for the majority of total complications (p b 0.005, OR 13.61, 95% CI (3.69, 50.20)). This met our predetermined level of significance (pb0.1), hence it was included in our model to control for its effects when determining the significance of the type of stoma on specific complications. Multiple-variable, logistic regression analysis was performed to determine the association between the type of stoma and the
Table 2 Stoma-related complications. Type of Complication
Loop
Divided
Total
p-value C
Retraction, n (%) Prolapse, n (%) Obstruction, n (%) Parastomal Hernia, n (%) Need for Stoma Revision, n (%) Megarectum, n (%) Urinary Tract Infection, n (%) Number of complications Number of patients who developed one or more complications
1 (1.4) 13 (17.8) 0 (0) 2 (3.0) 7 (9.6) 5 (8.2) 12 (16.4) 40 23 (31.5)
3 (4.2) 2 (2.8) 2 (2.8) 0 (0) 6 (8.4) 3 (5.3) 5 (7.0) 21 11 (15.5)
4 (2.8) 15 (10.4) 2 (1.4) 2 (1.5) 13 (9.0) 8 (6.8) 17 (11.8) 61 34 (23.6)
0.363 0.005⁎ 0.241 0.497 1.000 0.718 0.120 0.002⁎ 0.031⁎
C = Fisher’s Exact Test. ⁎ = Statistically significant value.
O. Oda et al. / Journal of Pediatric Surgery 49 (2014) 87–90 Table 3 Complications following anorectoplasty. Type of Complication
Loop
Divided
Total
P-valueC
Wound infection after anorectoplasty, n (%) Anal stricture, n (%) Fistula recurrence, n (%) Number of complications after anorectoplasty Number of patients who developed one or more complication after anorectoplasty, n (%)
9 (12.9)
4 (5.9)
13 (9.4)
0.244
4 (5.7) 2 (2.9) 15
6 (8.8) 1 (1.5) 11
10 (7.2) 3 (2.2) 26
0.529 1.000 0.739
13 (17.8)
9 (12.7)
22 (15.3)
0.487
89
Table 5 Multiple-variable logistic regression examining the association of of loop colostomies with the development of stoma related complications, controlling for the level of colostomy. Complication
Loop colostomies P-value
Retraction Prolapse obstruction Parastomal hernia Need for revision Megarectum UTI Patients with complication
C = Fisher’s Exact Test.
development of complications, controlling for effects of the level of the colostomy. The results are summarized in Table 5. Having a loop colostomy was found to be positively associated with the development of complications (p=0.033, OR 3.13, 95% CI (1.09, 8.96)) and stoma prolapse (p=0.009, OR 8.75, 95% CI (1.73, 44.16)), but not for any of the other complications. Neither the type nor the level of colostomy was associated with the development of complications following anorectoplasty and stoma closure.
3. Discussion Low types of ARMs can be repaired without a protective diverting colostomy. Although many reports have shown the safety of one stage corrective procedure for intermediate and high types of ARMs, a diverting colostomy is still considered the first step in the surgical management of these malformations [5,6]. By itself, creating a colostomy is a minor surgical procedure, but with potentially significant morbidity [7–9]. Complications include, but are not limited to, retraction, prolapse, parastomal hernia, bowel obstruction, skin excoriation, need for revision, and anastomotic leak and wound infections following stoma closure. The type of a diverting colostomy chosen depends on healthcare resources, surgeons’ training, personal experience and preference [10]. Some surgeons at our institution, and many other pediatric surgeons in the world, recommend a divided sigmoid colostomy in the left lower abdominal quadrant (LLQ) with a sufficient skin bridge between proximal stoma and distal mucous fistula that permits the appliance to be fitted on the proximal stoma allowing complete diversion of stool. They believe that complete stool diversion will prevent the development of megarectum, UTI, and wound infection after anorectoplasty [3,8]. Proponents of loop colostomy claim that a well-fashioned loop colostomy may not lead to such complications, and that a loop colostomy is easier to create and close; it also has the advantage of having better cosmetic results, particularly if it is fashioned at the site of umbilicus [11]. Some surgeons try to combine the alleged advantages of a divided stoma with the cosmetic advantage of umbilical incision by dividing the sigmoid colon and fashioning an end colostomy at the site of umbilicus and a mucus fistula in the LLQ. They claim that umbilical incision also allows for a better visualization of pelvic structures and
0.363 0.009⁎ 0.107 0.756 0.580 0.125 0.033⁎
OR
95%CI
8.75
1.74, 44.16
3.13
1.09, 8.96
OR: Odds Ratio. 95% CI: 95% Confidence Interval. - = Insufficient data to perform regression. ⁎ = Statistically significant value.
thus is a better option, particularly in complex ARMs and cloacas where additional procedures may be required. Although it is not mentioned in literature, it is the practice of many other surgeons to perform a double barrel sigmoid colostomy in the LLQ. These surgeons believe that disrupting bowel continuity is all that is required for complete stool diversion, and that a double barrel stoma can serve all purposes. The various types of stoma, the ongoing debate with regard to the best and least troublesome stoma, and the insufficient evidence in the literature created the basis for this study. Due to study limitations, including retrospective design and limited patient population (n=144), it was impractical to study each of the aforementioned stoma preferences separately. Thus, we divided all the patients into only two main groups: the first group included those who had a loop colostomy (where bowel wall continuity was partially preserved), and the second group included those who had a divided colostomy (where bowel continuity was completely disrupted regardless of distance between the proximal stoma and the distal mucous fistula). In summary, our results confirm the high incidence of stoma related complications in general, and the fact that sigmoid colostomies are more favorable than transverse colostomies. The results of our study also confirm that loop colostomies have a higher rate of complications than divided colostomies; this is principally related to prolapse. When other complications (retraction, obstruction, parastomal hernia, need for revision, post-anorectoplasty and post-stomaclosure wound infections) were studied separately, we found no difference between the two groups. Lastly, our results showed that development of a megarectum had no relation to the stoma type (loop vs divided). It is conceivable that a retracted loop colostomy could lead to stool accumulation in the distal pouch and the development of a megarectum. However, it is equally conceivable that a wellfashioned loop colostomy may not lead to such complications [8], which questions the need for bowel division and separation of bowel ends.
Table 4 Univariable logistic regression examining the association of the level of the colostomy with the development of stoma complications. Level of Colostomy
Development of a Complication
Sigmoid Descending Transverse
0 0.053⁎ 0.006⁎
OR
95%CI
p
OR: Odds Ratio. 95% CI: 95% Confidence Interval. ⁎ = statistically significant value.
Number of Complication
OR
95%CI
p 4.33
1.53, 12.24
0 0.607 0.047⁎
Prolapse
OR
95%CI
5.84 13.61
1.22, 27.78 3.69, 50.20
p 3.19
1.01, 10.03
0.027⁎ b0.005 ⁎
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O. Oda et al. / Journal of Pediatric Surgery 49 (2014) 87–90
Another controversy that was examined by the study was the association between the type of colostomy and the development of UTIs in patients with ARM. Although reports have shown no association between stoma type and UTIs in these patients [7], a common belief amongst pediatric surgeons is that loop colostomy can lead to a higher incidence of UTIs through the existing recto-urinary fistula [8]. Although our results showed a higher incidence of UTIs amongst those who had a loop colostomy (16.4% versus 7.0%), this was not statistically significant (p=0.120). Of interest was the additional finding that the majority of patients who developed UTIs had associated urinary tract anomalies or dysfunction, including neurogenic bladder and vesicourethral reflux. The presence of urinary tract anomalies or dysfunction was not initially a variable of interest in our study, thus it was not included in our analysis. However, in post-hoc review only one of the 17 patients who developed UTIs had no associated urinary tract anomalies or dysfunction; and 14 had UTI episodes after division of the recto-urinary fistula and repair of the ARM. A published collective review of 41 cases of epididymo-orchitis complicating ARM supports our findings[12]. According to the author, division of the recto-urinary fistula was curative only in one third of cases, which suggested that the presence of other risk factors, including persistent mesonephric duct syndrome, urethroejaculatory duct reflux, vasovesical ectopia, neurovesical dysfunction and urethral stricture-stenosis were the major risk factors for the development of epididymo-orchitis in those patients. We think that UTIs in patients with ARM are mainly the result of existing urinary tract anomalies or dysfunction, rather than the type of the diverting colostomy. The fact that there was no difference in the development of wound infections after anorectoplasty in our groups may indirectly support this point of view. 4. Conclusion Because of a higher incidence of prolapse, loop colostomies are associated with a higher total rate of complications than divided stomas. Other complications, including megarectum and UTIs are independent of stoma type. Generally speaking, the best type of the diverting sigmoid colostomy has not been defined yet. It remains the responsibility of the surgeon to determine if the benefits of cosmesis and easy closure warrant the increased risk of prolapse associated with loop colostomies. References [1] Yesildag E, Muniz RM. Buyukunal: How did the surgeons treat neonates with imperforate anus in the eighteenth century? Pediatr Surg Int 2010;26(12):1149–58. [2] Chandramouli B, Srinivasan K, Jagdish S, et al. Morbidity and Mortality of colostomy and its closure in children. J Pediatr Surg 2004;49(4):596–9. [3] Levitt MA, Kant A, Pena A. The morbidity of constipation in patients with anorectal malformations. J Pediatr Surg 2010;45(6):1228–33. [4] Peduzzi P, Concato J, Kemper E, et al. A simulation study of the number of events per variable in logistic regression analysis. J Clin Epidemiol 1996;49:1373–9. [5] Vick LR, Gosche JR, Boulanger SC, et al. Primary laparascopic repair of high imperforate anus in neonatal males. J Pediatr Surg 2007;42:1877–81. [6] Iwai N, Fumino S: Surgical treatment of anorectal malformations. Surg Today 2012 Nov 30 [Epub ahead of print]. [7] Patwardhan N, Kiely EM, Drake DP, et al. Colostomy for anorectal malformation: high incidence of complications. J Pediatr Surg 2001;36(5):795–8. [8] Pena A, Migatto-Krieger M, Levitt MA. Colostomy in anorectal malformations: a procedure with serious but preventable complications. J Pediatr Surg 2006;41(4): 748–55. [9] Chirdan LB, Uba FA, Ameh EA, et al. Colostomy for high anorectal malformation: an evaluation of morbidity in a developing country. Pediatr Surg Int 2008;24:407–10. [10] Chowdhary SK, Chalapathi G, Narasimhan KL, et al: An audit of neonatal colostomy for high anorectal malformation: the developing word perspective. [11] Hamada Y, Takada K, Nakamura Y, et al. Temporary umbilical loop colostomy for anorectal malformations. Pediatr Surg Int 2012;28:1133–6.
[12] Raveenthiran V, Sam CJ. Epididymo-orchitis complicating anorectal malformations: collective review of 41 cases. J Urol 2011;186(4):1467–72.
Discussion Discussant: Mr. Edward Kiely (London, UK): Can I ask you at what age did you close the stomas? How long were the stomas present? Sometimes if a loop stoma is present for many months prolapse is very common. If it is not present for many months, prolapse usually does not occur. Response: Dr Omar Oda: If you will look at this table, we have here the duration of the stoma in months. In the loop group it was eight months and in the divided group it was seven months. Mr. Edward kiely: I think if a loop stoma is present for less than three months you have no prolapse. It’s a very simple stoma to make if I can make a comment. It’s an easy stoma to close, and it is relatively trouble free if the reconstruction is to be done early. I don’t believe the stool comes out of the proximal stoma and looks around for the distal stoma to go burrowing down straight away. I don’t believe the distal stoma sucks it up either, so if you want to make a simple stoma and are going to close it soon, I have a strong preference for loop stoma. I see that Dr. Pena may be on his feet. (laughter). Moderater: Alberto has long been a champion of the divided colostomy. Dr. Pena, would you have a comment? Discussant: Dr Alberto Pena (Cincinnati, Oh): Yes. We studied in retrospect over 800 colostomies done in different parts of the world, so we have been exposed to all kinds of colostomies. Prolapse may occur in separated stomas or in loop colostomies. It doesn’t depend on the type of colostomy. We found that it depends on the location. If you open the colostomy in a mobile portion of the colon it most likely will prolapse regardless of the type that you perform. If you do it in a fixed portion of the colon, then it will not prolapse. And if you do it in a mobile portion of the colon, then you are obligated to fix the colon inside the abdomen in an adequate way. When you analyze urinary tract infections, of course it makes sense to believe that a loop colostomy allows the potential contamination of the urinary tract, but you have to separate the patients because there are patients that will have urinary tract infections for many other reasons. Patients that have tethered cord, absent sacrum, vesicoureteral reflux, cloacas we have to completely separate. Dr. Omar Oda: With respect, our data did not reveal any difference between urinary tract infections between the loop stoma and the divided stoma. When we analyzed our patients, those with recurrent UTIs, we found that 16 of the 17 had another urinary tract malformation, mainly vesicoureteral reflux which makes it more reasonable to infer that these urinary tract infections are related mainly to urinary tract malformation rather than the other issue. Of the 17 patients, 14 developed urinary tract infection even after repair of the malformation and division of the rectourinary fistula which suggests the rectourinary fistula may not have much to do with these infections. This has been reported also by other studies.
Contents lists available at ScienceDirect
Journal of Pediatric Surgery journal homepage: www.elsevier.com/locate/jpedsurg
Loop versus divided colostomy for the management of anorectal malformations Omar Oda, Dafydd Davies, Kimberly Colapinto, J. Ted Gerstle ⁎ Division of General and Thoracic Surgery, The Hospital for Sick Children, Toronto, ON, Canada
a r t i c l e
i n f o
Article history: Received 17 September 2013 Accepted 30 September 2013 Key words: Anorectal malformation Loop colostomy Divided colostomy
a b s t r a c t Purpose: The purpose of this study was to compare the clinical outcomes of loop and divided colostomies in patients with anorectal malformations (ARM). Methods: We performed a retrospective cohort study reviewing the medical records of all patients with ARM managed with diverting colostomies between 2000 and 2010 at our institution. Independent variables and outcomes of stoma complications were analyzed by parametric measures and logistic regression. Results: One hundred forty-four patients managed with a colostomy for ARM were evaluated (37.5% females, 50.7% loop, 49.3% divided). The incidence of patients with loop and divided colostomies who developed stoma-related complications was 31.5 and 15.5%, respectively (p=0.031). The incidence of prolapse was 17.8 and 2.8%, respectively (p=0.005). Multivariable-logistic regression controlling for other significant independent variables found loop colostomies to be positively associated with the development of a stoma complication (OR 3.13, 95%CI (1.09, 8.96), p=0.033). When individual complications were evaluated, it was only stoma prolapse that was more likely in patients with loop colostomies (OR 8.75, 95%CI (1.74, 44.16), p=0.009). Conclusion: Because of the higher incidence of prolapse, loop colostomies were found to be associated with a higher total incidence of complications than divided stomas. The development of other complications, including urinary tract infections (UTIs) and megarectum, were independent of the type of colostomy performed. © 2014 Elsevier Inc. All rights reserved.
Since the first diverting stoma performed for the treatment of imperforate anus in 1783 [1], the site and the type of the least troublesome stoma in the surgical management of anorectal malformations (ARM) have been major subjects for discussion amongst pediatric surgeons. Clinical studies have established the high morbidity associated with neonatal colostomy and that transverse colostomy has a higher rate of complications than sigmoid colostomy [2,3]. However debate continues regarding the type of the diverting colostomy. This study compares clinical outcomes of loop and divided colostomies performed as part of the surgical management of ARM. 1. Materials and methods Research Ethics Board approval (Application No: 1000031264, Protocol No: 1000020656) was obtained to perform a retrospective cohort study to review the medical records of all patients who presented with ARM and were managed with a diverting colostomy between November 2000 and November 2010. Patients with ARM who underwent primary corrective surgery without a diverting colostomy were not included in this study. Data were collected from ⁎ Corresponding author. Division of General and Thoracic Surgery, The Hospital for Sick Children, 555 University Avenue, Room 1526, Toronto, Ontario M5G 1X8, Canada. Tel.: +1 416 813 7500; fax: +1 416 813 7477. E-mail address: [email protected] (J.T. Gerstle). 0022-3468/$ – see front matter © 2014 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jpedsurg.2013.09.032
admission, progress, operative, discharge, and follow up clinic notes. Radiology reports were also reviewed. Demographic data collected included sex, type of ARM, and age at stoma creation. From the operative reports we determined whether a loop or divided colostomy was created. A loop colostomy was defined when the bowel wall continuity was partially preserved. A divided colostomy was defined when bowel continuity was completely disrupted regardless of the distance between the proximal and the distal ends of the stoma. For statistical purpose, stoma creation was considered early when age at stoma creation was less than 6 days, and late when age at stoma creation was equal or more than 6 days. The level of the colostomy (sigmoid, descending or transverse colon), the start of stoma function (days), and the period of time (months) the patient had the stoma were also determined. For those patients who had repair of the ARM at our institution, we noted whether a distal contrast study was performed prior to anorectoplasty and whether it showed the presence of a megarectum, as determined from the radiologist’s report. We determined if the patients developed complications from their stomas (for the time from stoma creation till stoma closure); these complications included retraction, prolapse, parastomal hernia, obstruction, and need for revision. The development of megarectum and UTIs were particular variables of interest; one of our goals was to establish if there was a relationship between them and the type of the colostomy, hence they were studied as potential stoma complications. For those who had repair of the ARM at
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our institution we documented if these patients developed any complications following anorectoplasty other than stoma complications. These included wound infection, anal stricture that required dilatation under general anesthesia or repeated anoplasty, and recurrence of recto-urinary or recto-genital fistula. A more detailed review of patients who developed UTIs was also performed; data included the presence of associated urinary tract malformations or dysfunction and wether these UTI episodes occurred before or after anorectoplasty and division of recto-urinary fistula. For those who had their stomas closed at our institution, we documented if they developed wound infections after closure of the colostomy. Statistical analysis was performed using STATA SE™ Version 10.0. The demographics and rates of complications were compared between patients initially managed with loop stomas to those managed with divided stomas. Categorical and continuous variables were analyzed with Fisher’s Exact and Wilcoxon Rank Sum Tests, respectively looking for differences between the two groups. Logistic regression analysis was performed. Along with the type of colostomy (loop versus divided), independent variables of sex, type of ARM, age at stoma creation, level of stoma, and duration of stoma were evaluated for associations with the outcomes of stoma complications (retraction, prolapse, parastomal hernia, obstruction, need for stoma revision, and the development of megarectum and UTIs). We also examined their impact on complications following ARM repair and stoma closure including wound infection, development of anal stricture or recurrent fistula. In order to prevent the inclusion of variables with no outcome association in our multiple variable logistic regression analysis, only independent variables with associations meeting significance levels of p less that 0.1 were included to control for their effects. This was repeated for each outcome measure if the number of events was sufficient [4]. The results of the multiple variable logistic regression analysis were only considered significant if they reached a p-value less than 0.05. 2. Results Over the 10 year study period 461 children with all types of ARM (low, intermediate, and high) were treated at our institution. 317 patients underwent a primary repair of the ARM without a colostomy and were excluded from the study. 144 patients were managed with a colostomy. 73 (50.7%) patients had a loop colostomy, and 71 (49.3%) patients had a divided colostomy. 112 (77.7%) had sigmoid colostomy, 14 (9.7%) had descending colostomy, and 18 (12.5%) had transverse colostomy. 138 (95.8%) patients underwent anorectoplasty at our institution: 112 underwent a posterior saggital anorectoplasty (PSARP), 18 underwent a laparascopic assisted anorectoplasty, and in 7 patients anorectoplasty was performed with an open abdominoperineal approach. The operative report was not found for one patient and we were unable to determine what type of anorectoplasty he had, despite it being performed at our institution. These, and the demographic data are shown in Table 1. 118 (85.5%) of repaired patients underwent a distal contrast study prior to anorectoplasty, and 130 (94.2%) had their colostomies closed at our institution. With respect to demographics and other data shown in table 1 including age at stoma creation (early vs. delayed), there were no statistically significant differences between patients who had a loop and those who had a divided colostomy. Stoma related complications occurred in 34 (23.6%) patients (some with more than one) giving a total of 61 complications in 144 patients. The types and rates of complications are summarized in Table 2. Patients with loop stomas were significantly more likely to develop complications (p=0.031) and the number of complications was higher in this group (0.002). When comparing the rates of developing each individual complication, only the rate of stoma prolapse was found to be statistically higher in patients with loop stomas (p=0.005).
Table 1 Demographics. Variable
Loop
n (%) Sex Male, n (%) Type of Anorectal Malformation Perineal Fistula, n (%) Vestibular Fistula, n (%) Cloaca, n (%) Recto-urethral Fistula, n (%) Recto-Bladder Neck Fistula, n (%) H-Type Fistula, n (%) Atresia without Fistula, n (%) Level of Colostomy Sigmoid, n (%) Descending, n (%) Transverse, n (%) Age at Colostomy, median (IQR) (days) Delayed Stoma Creation N 6 days old, n (%) Duration of Stoma, months (n=130), median (IQR) Type of Anorectoplasty (n=138) PSARP, n (%) Laparoscopic Assisted anorectoplasty, n (%) Abdominoperineal Approach, n (%) Undefined
73 (50.7) 71 (49.3) 144 (100%) 42 (46.7) 48 (53.3) 90 (62.5) 0.232 0.600 4 (5.5) 8 (11.3) 12 (8.3) 16 (22.0) 11 (15.5) 27 (18.8) 7 (9.6) 8 (11.3) 15 (10.4) 26 (35.6) 22 (31.0) 48 (33.3) 6 (8.2) 7 (9.9) 13 (9) 2 (2.7) 0 (0) 2 (1.4) 12 (16.4) 15 (21.1) 27 (18.8) 0.615 56 (76.7) 56 (78.9) 112 (77.8) 6 (8.2) 8 (11.3) 14 (9.7) 11 (15.1) 7 (9.9) 18 (12.5) 2 (1, 3) 2 (1, 3) 2 (1, 2) 0.914
Divided
Total
p-value C C
C
⩖
17 (22.3) 13 (18.3) 30 (20.8)
0.540 C
8 (6, 10)
0.085 ⩖
7 (5, 10)
8 (6, 10)
0.091 C 61 (87.1) 51 (75.0) 112 5 (7.1) 13 (19.1) 18 3 (4.3) 1 (1.4)
4 (5.9) 0 (0)
7 1
C = Fisher’s Exact test. ⩖ = Wilcoxon Rank Sum Test.
Anorectoplasty was performed at our institution on 138 patients. There were a total of 26 complications following anorectoplasty which occurred in 22 (15.3%) patients. These are summarized in Table 3 and were no more frequent in either group. The results of the univariable logistic regression found that sex, type of ARM, age at stoma creation, and duration of the stoma were neither positively associated with the development of complications nor the development of any of the specific complications evaluated; having not met our predetermined level of significance (p=0.1) they were not included in the multivariable analysis. The level of colostomy was found to be positively associated with the development of stoma complications. As shown in Table 4, sigmoid colostomies were most favorable, whereas transverse colostomies had the highest association with the development of complications (p=0.006, OR 4.33, 95% CI (1.53, 12.24)). Again, prolapse accounted for the majority of total complications (p b 0.005, OR 13.61, 95% CI (3.69, 50.20)). This met our predetermined level of significance (pb0.1), hence it was included in our model to control for its effects when determining the significance of the type of stoma on specific complications. Multiple-variable, logistic regression analysis was performed to determine the association between the type of stoma and the
Table 2 Stoma-related complications. Type of Complication
Loop
Divided
Total
p-value C
Retraction, n (%) Prolapse, n (%) Obstruction, n (%) Parastomal Hernia, n (%) Need for Stoma Revision, n (%) Megarectum, n (%) Urinary Tract Infection, n (%) Number of complications Number of patients who developed one or more complications
1 (1.4) 13 (17.8) 0 (0) 2 (3.0) 7 (9.6) 5 (8.2) 12 (16.4) 40 23 (31.5)
3 (4.2) 2 (2.8) 2 (2.8) 0 (0) 6 (8.4) 3 (5.3) 5 (7.0) 21 11 (15.5)
4 (2.8) 15 (10.4) 2 (1.4) 2 (1.5) 13 (9.0) 8 (6.8) 17 (11.8) 61 34 (23.6)
0.363 0.005⁎ 0.241 0.497 1.000 0.718 0.120 0.002⁎ 0.031⁎
C = Fisher’s Exact Test. ⁎ = Statistically significant value.
O. Oda et al. / Journal of Pediatric Surgery 49 (2014) 87–90 Table 3 Complications following anorectoplasty. Type of Complication
Loop
Divided
Total
P-valueC
Wound infection after anorectoplasty, n (%) Anal stricture, n (%) Fistula recurrence, n (%) Number of complications after anorectoplasty Number of patients who developed one or more complication after anorectoplasty, n (%)
9 (12.9)
4 (5.9)
13 (9.4)
0.244
4 (5.7) 2 (2.9) 15
6 (8.8) 1 (1.5) 11
10 (7.2) 3 (2.2) 26
0.529 1.000 0.739
13 (17.8)
9 (12.7)
22 (15.3)
0.487
89
Table 5 Multiple-variable logistic regression examining the association of of loop colostomies with the development of stoma related complications, controlling for the level of colostomy. Complication
Loop colostomies P-value
Retraction Prolapse obstruction Parastomal hernia Need for revision Megarectum UTI Patients with complication
C = Fisher’s Exact Test.
development of complications, controlling for effects of the level of the colostomy. The results are summarized in Table 5. Having a loop colostomy was found to be positively associated with the development of complications (p=0.033, OR 3.13, 95% CI (1.09, 8.96)) and stoma prolapse (p=0.009, OR 8.75, 95% CI (1.73, 44.16)), but not for any of the other complications. Neither the type nor the level of colostomy was associated with the development of complications following anorectoplasty and stoma closure.
3. Discussion Low types of ARMs can be repaired without a protective diverting colostomy. Although many reports have shown the safety of one stage corrective procedure for intermediate and high types of ARMs, a diverting colostomy is still considered the first step in the surgical management of these malformations [5,6]. By itself, creating a colostomy is a minor surgical procedure, but with potentially significant morbidity [7–9]. Complications include, but are not limited to, retraction, prolapse, parastomal hernia, bowel obstruction, skin excoriation, need for revision, and anastomotic leak and wound infections following stoma closure. The type of a diverting colostomy chosen depends on healthcare resources, surgeons’ training, personal experience and preference [10]. Some surgeons at our institution, and many other pediatric surgeons in the world, recommend a divided sigmoid colostomy in the left lower abdominal quadrant (LLQ) with a sufficient skin bridge between proximal stoma and distal mucous fistula that permits the appliance to be fitted on the proximal stoma allowing complete diversion of stool. They believe that complete stool diversion will prevent the development of megarectum, UTI, and wound infection after anorectoplasty [3,8]. Proponents of loop colostomy claim that a well-fashioned loop colostomy may not lead to such complications, and that a loop colostomy is easier to create and close; it also has the advantage of having better cosmetic results, particularly if it is fashioned at the site of umbilicus [11]. Some surgeons try to combine the alleged advantages of a divided stoma with the cosmetic advantage of umbilical incision by dividing the sigmoid colon and fashioning an end colostomy at the site of umbilicus and a mucus fistula in the LLQ. They claim that umbilical incision also allows for a better visualization of pelvic structures and
0.363 0.009⁎ 0.107 0.756 0.580 0.125 0.033⁎
OR
95%CI
8.75
1.74, 44.16
3.13
1.09, 8.96
OR: Odds Ratio. 95% CI: 95% Confidence Interval. - = Insufficient data to perform regression. ⁎ = Statistically significant value.
thus is a better option, particularly in complex ARMs and cloacas where additional procedures may be required. Although it is not mentioned in literature, it is the practice of many other surgeons to perform a double barrel sigmoid colostomy in the LLQ. These surgeons believe that disrupting bowel continuity is all that is required for complete stool diversion, and that a double barrel stoma can serve all purposes. The various types of stoma, the ongoing debate with regard to the best and least troublesome stoma, and the insufficient evidence in the literature created the basis for this study. Due to study limitations, including retrospective design and limited patient population (n=144), it was impractical to study each of the aforementioned stoma preferences separately. Thus, we divided all the patients into only two main groups: the first group included those who had a loop colostomy (where bowel wall continuity was partially preserved), and the second group included those who had a divided colostomy (where bowel continuity was completely disrupted regardless of distance between the proximal stoma and the distal mucous fistula). In summary, our results confirm the high incidence of stoma related complications in general, and the fact that sigmoid colostomies are more favorable than transverse colostomies. The results of our study also confirm that loop colostomies have a higher rate of complications than divided colostomies; this is principally related to prolapse. When other complications (retraction, obstruction, parastomal hernia, need for revision, post-anorectoplasty and post-stomaclosure wound infections) were studied separately, we found no difference between the two groups. Lastly, our results showed that development of a megarectum had no relation to the stoma type (loop vs divided). It is conceivable that a retracted loop colostomy could lead to stool accumulation in the distal pouch and the development of a megarectum. However, it is equally conceivable that a wellfashioned loop colostomy may not lead to such complications [8], which questions the need for bowel division and separation of bowel ends.
Table 4 Univariable logistic regression examining the association of the level of the colostomy with the development of stoma complications. Level of Colostomy
Development of a Complication
Sigmoid Descending Transverse
0 0.053⁎ 0.006⁎
OR
95%CI
p
OR: Odds Ratio. 95% CI: 95% Confidence Interval. ⁎ = statistically significant value.
Number of Complication
OR
95%CI
p 4.33
1.53, 12.24
0 0.607 0.047⁎
Prolapse
OR
95%CI
5.84 13.61
1.22, 27.78 3.69, 50.20
p 3.19
1.01, 10.03
0.027⁎ b0.005 ⁎
90
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Another controversy that was examined by the study was the association between the type of colostomy and the development of UTIs in patients with ARM. Although reports have shown no association between stoma type and UTIs in these patients [7], a common belief amongst pediatric surgeons is that loop colostomy can lead to a higher incidence of UTIs through the existing recto-urinary fistula [8]. Although our results showed a higher incidence of UTIs amongst those who had a loop colostomy (16.4% versus 7.0%), this was not statistically significant (p=0.120). Of interest was the additional finding that the majority of patients who developed UTIs had associated urinary tract anomalies or dysfunction, including neurogenic bladder and vesicourethral reflux. The presence of urinary tract anomalies or dysfunction was not initially a variable of interest in our study, thus it was not included in our analysis. However, in post-hoc review only one of the 17 patients who developed UTIs had no associated urinary tract anomalies or dysfunction; and 14 had UTI episodes after division of the recto-urinary fistula and repair of the ARM. A published collective review of 41 cases of epididymo-orchitis complicating ARM supports our findings[12]. According to the author, division of the recto-urinary fistula was curative only in one third of cases, which suggested that the presence of other risk factors, including persistent mesonephric duct syndrome, urethroejaculatory duct reflux, vasovesical ectopia, neurovesical dysfunction and urethral stricture-stenosis were the major risk factors for the development of epididymo-orchitis in those patients. We think that UTIs in patients with ARM are mainly the result of existing urinary tract anomalies or dysfunction, rather than the type of the diverting colostomy. The fact that there was no difference in the development of wound infections after anorectoplasty in our groups may indirectly support this point of view. 4. Conclusion Because of a higher incidence of prolapse, loop colostomies are associated with a higher total rate of complications than divided stomas. Other complications, including megarectum and UTIs are independent of stoma type. Generally speaking, the best type of the diverting sigmoid colostomy has not been defined yet. It remains the responsibility of the surgeon to determine if the benefits of cosmesis and easy closure warrant the increased risk of prolapse associated with loop colostomies. References [1] Yesildag E, Muniz RM. Buyukunal: How did the surgeons treat neonates with imperforate anus in the eighteenth century? Pediatr Surg Int 2010;26(12):1149–58. [2] Chandramouli B, Srinivasan K, Jagdish S, et al. Morbidity and Mortality of colostomy and its closure in children. J Pediatr Surg 2004;49(4):596–9. [3] Levitt MA, Kant A, Pena A. The morbidity of constipation in patients with anorectal malformations. J Pediatr Surg 2010;45(6):1228–33. [4] Peduzzi P, Concato J, Kemper E, et al. A simulation study of the number of events per variable in logistic regression analysis. J Clin Epidemiol 1996;49:1373–9. [5] Vick LR, Gosche JR, Boulanger SC, et al. Primary laparascopic repair of high imperforate anus in neonatal males. J Pediatr Surg 2007;42:1877–81. [6] Iwai N, Fumino S: Surgical treatment of anorectal malformations. Surg Today 2012 Nov 30 [Epub ahead of print]. [7] Patwardhan N, Kiely EM, Drake DP, et al. Colostomy for anorectal malformation: high incidence of complications. J Pediatr Surg 2001;36(5):795–8. [8] Pena A, Migatto-Krieger M, Levitt MA. Colostomy in anorectal malformations: a procedure with serious but preventable complications. J Pediatr Surg 2006;41(4): 748–55. [9] Chirdan LB, Uba FA, Ameh EA, et al. Colostomy for high anorectal malformation: an evaluation of morbidity in a developing country. Pediatr Surg Int 2008;24:407–10. [10] Chowdhary SK, Chalapathi G, Narasimhan KL, et al: An audit of neonatal colostomy for high anorectal malformation: the developing word perspective. [11] Hamada Y, Takada K, Nakamura Y, et al. Temporary umbilical loop colostomy for anorectal malformations. Pediatr Surg Int 2012;28:1133–6.
[12] Raveenthiran V, Sam CJ. Epididymo-orchitis complicating anorectal malformations: collective review of 41 cases. J Urol 2011;186(4):1467–72.
Discussion Discussant: Mr. Edward Kiely (London, UK): Can I ask you at what age did you close the stomas? How long were the stomas present? Sometimes if a loop stoma is present for many months prolapse is very common. If it is not present for many months, prolapse usually does not occur. Response: Dr Omar Oda: If you will look at this table, we have here the duration of the stoma in months. In the loop group it was eight months and in the divided group it was seven months. Mr. Edward kiely: I think if a loop stoma is present for less than three months you have no prolapse. It’s a very simple stoma to make if I can make a comment. It’s an easy stoma to close, and it is relatively trouble free if the reconstruction is to be done early. I don’t believe the stool comes out of the proximal stoma and looks around for the distal stoma to go burrowing down straight away. I don’t believe the distal stoma sucks it up either, so if you want to make a simple stoma and are going to close it soon, I have a strong preference for loop stoma. I see that Dr. Pena may be on his feet. (laughter). Moderater: Alberto has long been a champion of the divided colostomy. Dr. Pena, would you have a comment? Discussant: Dr Alberto Pena (Cincinnati, Oh): Yes. We studied in retrospect over 800 colostomies done in different parts of the world, so we have been exposed to all kinds of colostomies. Prolapse may occur in separated stomas or in loop colostomies. It doesn’t depend on the type of colostomy. We found that it depends on the location. If you open the colostomy in a mobile portion of the colon it most likely will prolapse regardless of the type that you perform. If you do it in a fixed portion of the colon, then it will not prolapse. And if you do it in a mobile portion of the colon, then you are obligated to fix the colon inside the abdomen in an adequate way. When you analyze urinary tract infections, of course it makes sense to believe that a loop colostomy allows the potential contamination of the urinary tract, but you have to separate the patients because there are patients that will have urinary tract infections for many other reasons. Patients that have tethered cord, absent sacrum, vesicoureteral reflux, cloacas we have to completely separate. Dr. Omar Oda: With respect, our data did not reveal any difference between urinary tract infections between the loop stoma and the divided stoma. When we analyzed our patients, those with recurrent UTIs, we found that 16 of the 17 had another urinary tract malformation, mainly vesicoureteral reflux which makes it more reasonable to infer that these urinary tract infections are related mainly to urinary tract malformation rather than the other issue. Of the 17 patients, 14 developed urinary tract infection even after repair of the malformation and division of the rectourinary fistula which suggests the rectourinary fistula may not have much to do with these infections. This has been reported also by other studies.