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DOI: 10.1055/s-0033-1343085
Open Versus Transanal Pull-Through for Hirschsprung Disease: A Systematic Review of Long-Term Outcome
Address for correspondence
Publication History
12 March 2013
12 March 2013
Publication Date:
09 April 2013 (online)
- Introduction
- Methods
- Results
- Discussion
- Drawbacks/Limitations of the Study
- Conclusion
- References
Abstract
Introduction Various surgical techniques for the treatment of Hirschsprung disease (HD) have been proposed. The most relevant long-term complications of HD surgery include constipation, soiling/incontinence, enterocolitis, and anastomotic stricture. To date, there has been no randomized controlled trial evaluating the long-term outcome of OPEN surgery compared with transanal approaches with and without laparoscopy (laparoscopic-assisted transanal-endorectal pull-through [L-TERPT] and transanal-endorectal pull-through [TERPT]). We performed a systematic literature review of the long-term outcome of OPEN surgery compared with L-TERPT/TERPT.
Methods Our systematic review of the recent literature (2008 to 2012) included reports on long-term outcome of either OPEN surgery or L-TERPT/TERPT with a minimum follow-up period of 12 months. With the cumulative data, a comparative meta-analysis was performed for the outcome parameters “constipation,” “soiling/incontinence,” “enterocolitis,” and “anastomotic stricture.”
Results Functional outcome of surgical techniques for HD was highly variable. We could show a significant advantage of L-TERPT/TERPT over OPEN surgery regarding the incidence of soiling/incontinence and constipation. No differences were seen for enterocolitis and anastomotic stricture.
Conclusion Significant differences in the long-term outcome of OPEN surgery compared with L-TERPT/TERPT were identified in this review. We conclude from our data that L-TERPT/TERPT represents a valid option in the treatment of HD and might have some advantages over the OPEN techniques. However, the present data should be interpreted carefully due to limitations in the quality of the study design in most reports. Prospective, randomized, multicenter trials are urgently needed to overcome this weakness of the current literature.
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Introduction
Various surgical procedures for the treatment of Hirschsprung disease (HD) have been proposed. The principle of all techniques is the resection of the aganglionic segment and anastomosis. In 1948, Swenson and Bill first described a staged repair for neonates and infants with HD: preliminary colostomy followed by a pull-through operation via laparotomy.[1] In the early 1980s, a better understanding of disease pathogenesis with earlier diagnosis together with technical refinements allowed surgeons to perform single-stage pull-throughs.[2]
The next milestone was reached in the 1990s, when Georgeson et al minimized the invasiveness of the procedure by performing a transanal mucosectomy combined with a laparoscopic bowel resection.[3] In addition to all the benefits of a minimally invasive technique, the advantage of this operation was the possibility to obtain intraoperative seromuscular biopsies to determine the extension of aganglionosis before starting endorectal dissection and mesenteric division. The introduction of a single-stage transanal endorectal pull-through (TERPT) by de la Torre-Mondragon and Ortega in 1998[4] drastically changed the treatment of HD. This operation was performed entirely via the anus and did not require a laparotomy or laparoscopy, therefore potentially minimizing the risk of intraperitoneal contamination, adhesion formation, and damaging the pelvic structures. As it offered not only the safety and efficacy of the previous techniques but also all the advantages of a minimally invasive technique such as better pain control, faster discharge from hospital, and a better cosmetic result, this technique became the most popular approach for the treatment of HD in many institutions.[5] [6] [7] [8]
In the last decade, it has been well documented that TERPT is a safe and efficient operation with early complication rates comparable to laparoscopic or open procedures.[7] [9] [10] However, the possible consequences of transanal mobilization of rectum and colon by stretching the anal sphincters have caused considerable concern about long-term anorectal function, particularly soiling, constipation, and incontinence.[10] [11] [12] [13] [14]
Therefore, the aim of this study was to review the available data on long-term functional results in HD patients operated with conventional transabdominal pull-through (OPEN) compared with transanal approaches with and without laparoscopy (laparoscopic-assisted transanal-endorectal pull-through [L-TERPT] and transanal-endorectal pull-through [TERPT]) reported in the recent literature.
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Methods
A systematic PubMed search was performed to identify publications on outcome of children with HD, who underwent either OPEN, L-TERPT, or TERPT. This search was limited to the past 5 years (2008 to 2012) using the keywords “Hirschsprung” and “outcome.” Reference lists were screened for additional studies.
Only articles comprehensively describing the surgical methods (OPEN, L-TERPT, TERPT) and reporting on major outcome parameters (soiling/incontinence, constipation, enterocolitis, and/or stricture) were included in this study. Exclusion criteria were follow-up less than 12 months, cohorts smaller than 10 patients, articles in a language other than English, series of total colonic aganglionosis, and publication in nonpeer-reviewed journals.
Data extracted from included articles were study characteristics (authors, journal, publication year, sample size, surgical technique, and follow-up period) and patient characteristics (age at surgery, extent of aganglionosis). Specific information on the outcome parameters constipation, enterocolitis, stricture, incontinence, and soiling were collected and analyzed.
Since not all outcome parameters were reported in all articles, each specific parameter was analyzed according to the number of patients in whom that item was assessed. This enabled the calculation of the incidence of each specific outcome parameter. With these data, a cumulative meta-analysis was performed to compare long-term outcome of patients who underwent OPEN surgery with patients who underwent L-TERPT/TERPT surgery. The latter groups were analyzed as one group due to the transanal approach in both. Odds ratio (OR) with 95% confidence interval (CI) were calculated. Statistical differences were considered as significant for a p value < 0.05 (two-tailed).
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Results
The initial literature search resulted in a total number of 148 articles published from 2008 to 2012. Of these, 130 articles were excluded because they did not fulfill the inclusion criteria ([Fig. 1]). Eighteen articles finally met the inclusion criteria and were analyzed in detail.[10] [14] [15] [16] [17] [18] [19] [20] [21] [22] [23] [24] [25] [26] [27] [28] [29] [30]
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Study Population Characteristics
A total of 1,412 subjects with HD were included in this review (range: 10 to 192 patients per study). Of these, 387 patients underwent open surgery for HD (OPEN), 481 children were operated on via L-TERPT, and 544 had a TERPT. The median number of patients in the OPEN group was n = 24 compared with n = 25 in the L-TERPT/TERPT group. Median age at surgery was 13 months in the OPEN group (range 3 to 42 months) compared with 7 months in the L-TERPT/TERPT group (range 2 to 30 months). Statistical analysis of group sizes and age at surgery revealed no significant difference (p > 0.05). Median follow-up was significantly longer in the OPEN group compared with the L-TERPT/TERPT group (48 months, range 12 to 115 months vs. 36 months, range 12 to 80 months; p < 0.05).
Constipation
The mean rate of constipation was 21% (range 0 to 59%) in the OPEN group compared with 10% (range 0 to 27%) in the L-TERPT/TERPT group. A cumulative meta-analysis revealed that constipation was significantly more frequent after OPEN surgery compared with L-TERPT/TERPT (OR 3.89 [95% CI, 2.69 to 5.62]; p < 0.0001) ([Fig. 2], [Table 1]).
Abbreviations: CI, confidence interval; L-TERPT, transanal pull-through with laparoscopic assistance; OPEN, conventional pull-through procedure via laparotomy; OR, odds ratio; TERPT, transanal pull-through without laparoscopic assistance.
Note: Constipation was significantly more frequent after OPEN surgery compared with L-TERPT/TERPT (OR 3.89 [95% CI, 2.69 to 5.62]; p < 0.0001).
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Soiling/Incontinence
The mean rate of soiling/incontinence was 33% (range 10 to 75%) in the OPEN group compared with 25% (range 2 to 88%) in the L-TERPT/TERPT group. Soiling/incontinence occurred significantly more frequently after OPEN surgery compared with L-TERPT/TERPT (OR 3.87 [95% CI, 2.83 to 5.31]; p < 0.0001) ([Fig. 2], [Table 2]).
Abbreviations: CI, confidence interval; L-TERPT, transanal pull-through with laparoscopic assistance; OPEN, conventional pull-through procedure via laparotomy; OR, odds ratio; TERPT, transanal pull-through without laparoscopic assistance.
Note: Soiling/incontinence occurred significantly more frequently after OPEN surgery compared with L-TERPT/TERPT (OR 3.87[95% CI, 2.83 to 5.31]; p < 0.0001).
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Enterocolitis
The mean frequency of enterocolitis was 14% (range 3 to 31%) in the OPEN group compared with 14% (range 0 to 34%) in the L-TERPT/TERPT group. There was no significant difference between the rate of enterocolitis after OPEN pull-through and the rate of enterocolitis after L-TERPT/TERPT (OR 1.04 [95% CI, 0.73 to 1.50]; p = 0.8177) ([Fig. 2], [Table 3]).
Abbreviations: CI, confidence interval; L-TERPT, transanal pull-through with laparoscopic assistance; OPEN, conventional pull-through procedure via laparotomy; OR, odds ratio; TERPT, transanal pull-through without laparoscopic assistance.
Note: There was no significant difference between the rate of enterocolitis after OPEN pull-through and the rate of enterocolitis after L-TERPT/TERPT (OR 1.04 [95% CI, 0.73 to 1.50]; p = 0.8177).
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Anastomotic Stricture
Seven percent (range 0 to 21%) of children in the OPEN group developed an anastomotic stricture compared with 9% (range 0 to 43%) in the L-TERPT/TERPT group. No significant difference was found comparing occurrence of strictures after OPEN pull-through versus L-TERPT/TERPT (OR 1.16 [95% CI, 0.66 to 2.04]; p = 0.5989) ([Fig. 2], [Table 4]).
Abbreviations: CI, confidence interval; L-TERPT, transanal pull-through with laparoscopic assistance; OPEN, conventional pull-through procedure via laparotomy; OR, odds ratio; TERPT, transanal pull-through without laparoscopic assistance.
Note: No significant difference was found comparing occurrence of strictures after OPEN pull-through versus L-TERPT/TERPT (OR 1.16 [95% CI, 0.66 to 2.04]; p = 0.5989).
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Discussion
Several large multicenter series confirmed an excellent safety and feasibility of L-TERPT/TERPT. It was suggested that L-TERPT and TERPT had advantages over OPEN procedures such as shorter time to feeding, shorter hospital stay, less pain, and better cosmesis.[15] [31] [32] [33] [34] [35] Recently, Langer et al discussed that although L-TERPT and TERPT are often thought of as separate approaches, the techniques are similar in most aspects.[33] The main difference is the preparation of the rectum: Laparoscopy allows additional dissection of the rectum from above, whereas by using a transanal technique, the rectum is dissected from below only. The authors further stated that many “transanal” surgeons used laparoscopy for biopsy and/or mobilization of the proximal colon without using it for rectal dissection, making the “L-TERPT” technique a very heterogeneous approach. Therefore, no study has been published to date comparing L-TERPT with TERPT and Langer conclude that this question has increasingly become less relevant.[33] Hence, for the present study, we decided to analyze L-TERPT and TERPT as one group.
However, functional results after transanal repair versus open techniques may not be superior and even associated with additional problems due to extensive stretching by retractors to gain visualization with subsequent damage to the anal canal.[36]
To date, there has been no randomized controlled study evaluating the long-term outcome of OPEN surgery compared with L-TERPT/TERPT.[36] [37] Therefore, the present review could only focus on retrospective studies with a high variability of follow-up periods and definition of outcome parameters. We focused on long-term outcome in HD patients operated with conventional transabdominal pull-through (OPEN) compared with transanal endorectal pull-through with or without laparoscopy (L-TERPT/TERPT) using four major outcome parameters: constipation, incontinence, enterocolitis and anastomotic stricture.
Constipation
The incidence of postpull-through constipation in the current literature ranges from 0 to 59%.[10] [20] [38] [39] Suspected causes include abnormal motility of the pulled-through segment and abnormal function of the internal anal sphincter due to the underlying disease or preceding surgery.[36] [37] [40] However, most authors describe an improvement of constipation in late childhood. Our systematic review revealed postoperative constipation rates of up to 59%. Furthermore, it confirms previous reports on a higher incidence of postoperative constipation after OPEN surgery when compared with L-TERPT/TERPT.[39] [41] [42] The causes for these findings remain elusive. However, one could speculate that extensive pelvic dissection as it is performed in OPEN surgery may lead to iatrogenic pelvic-nerve injury compromising rectal motility and subsequently leading to constipation.[33] [36] [37] [43]
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Soiling/Incontinence
Most patients with HD have been reported to suffer from soiling which remains the major cause of psychological and social problems in these patients.[40] [44] Frank incontinence is rare. Besides constipation, fecal soiling/incontinence has therefore been identified as the most relevant long-term complication after any type of surgery for HD.[36] [40] [45] [46] [47] [48] Three major causes of soiling have been described: abnormal sphincter function, abnormal sensation, and “pseudoincontinence.” The latter is caused by either severe constipation with fecal overflow or hypermotility of the pulled-through colon.[33] [36] [37] Levitt et al postulate that sphincter function and sensation may be damaged during the primary repair, and that the colonic motility is substantially affected by an operation for HD.[36] However, it is noteworthy that patients with HD lack rectoanal relaxation reflex not only after but also before surgery.[40] [49] [50] Therefore, it remains unclear to what extent surgical dissection of the rectum compromises relevant nerval structures responsible for anal canal sensation, sphincter competence, and colonic motility.
There is an ongoing debate on whether the incidence of soiling/incontinence is associated with the operative technique.[40] [51] [52] [53] Some authors suggest that extensive mechanical stretching during L-TERPT/TERPT by retractors can damage the anal canal and might therefore lead to a higher incidence of incontinent HD patients.[36] However, our data do not reflect these concerns as it shows that soiling/incontinence occurs significantly more often after OPEN compared with L-TERPT/TERPT surgery. The higher incidence of soiling/incontinence in the OPEN surgery group may derive from deep pelvic dissection affecting pelvic nerves and therefore compromising both sphincter function and sensation.[33] On the other hand, the frequency of constipation was also higher in the OPEN group which could indicate overflow incontinence.
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Enterocolitis
Hirschsprung-associated enterocolitis (HAEC) represents another major postoperative complication and is the most common cause of death in children with HD.[33] [54] The pathogenesis of HAEC is still unknown.[39] [55] Several risk factors for HAEC have been identified including diagnosis at young age, anastomotic stricture, and malnutrition.[39] [56] [57]
Some authors report that obstruction at the level of the anus leading to intestinal stasis may play a major role in HAEC pathogenesis.[39] [58] The potential reasons for intestinal stasis include a spastic or long cuff,[39] [59] presuming that a shorter cuff might help to reduce postoperative enterocolitis.[39] [59] However, as HAEC may occur both before and after surgery for HD,[60] the high incidence of HAEC cannot be explained by partial obstruction alone.[39]
We did not find a significant difference in the incidence of enterocolitis after OPEN surgery compared with L-TERPT/TERPT. This may indicate that HAEC is rather part of the underlying pathology of HD than a result of a particular surgical technique. Moreover, the lack of a clear definition of HAEC remains a major problem that could account for the wide range of incidence reported from 5 to 50%.[60]
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Anastomotic Stricture
Multiple risk factors for anastomotic stricture including anastomotic ischemia, anastomotic leakage, and cuff ischemia have been described.[39] [41] [61] Various surgical techniques were suggested to prevent stricture formation, such as an oblique coloanal anastomosis reported by Swenson.[39] [62]
The studies analyzed in this review showed no significant difference in the incidence of anastomotic stricture after OPEN surgery compared with L-TERPT/TERPT surgery.
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Drawbacks/Limitations of the Study
Although the present review of the recent literature revealed a higher incidence of constipation and soiling/incontinence after OPEN surgery compared with L-TERPT/TERPT, these data need to be carefully interpreted. The following limitations of the present analysis and validity of the data on the long-term outcome in the current literature may be discussed:
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There was a lack of uniform definitions of outcome parameters after surgery for HD such as constipation, soiling, incontinence, enterocolitis, and stricture.
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In a very few studies, standardized scores to describe endpoints were used.
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L-TERPT and TERPT were analyzed as one group, which is due to the difficulty to distinguish whether laparoscopy was used for biopsies only or for dissection of the rectum. A comprehensive description of the surgical technique was lacking in numerous reports.
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The OPEN surgery group consisted of several pull-through techniques such as Soave, Swenson, Duhamel, and Rehbein which might have led to a high variability of results.
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The OPEN group was comparable to the L-TERPT/TERPT group in terms of “age at surgery” and “group size” but the follow-up period was significantly longer. As the functional results tend to be more favorable with increasing age, the high rate of constipation and soiling in the OPEN group is remarkable.
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Comparative, randomized studies evaluating outcome of OPEN surgery versus L-TERPT/TERPT were not available for this review. Therefore, the validity of the data and level of evidence of the present analysis is limited.
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Conclusion
Functional outcome of different surgical techniques for HD is highly variable. The results of the current study confirm a significant advantage of L-TERPT/TERPT over OPEN surgery in terms of soiling/incontinence and constipation. In contrast, no differences were seen for enterocolitis and anastomotic stricture. Several limitations to this systematic review such as the different quality of studies and small cohort sizes need to be considered when interpreting these data. Moreover, only few authors used standardized scores to assess functional outcome. Hence, prospective, randomized, multicenter trials are urgently needed to overcome this weakness of the available reports in current literature.
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Conflict of Interest
None
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- 59 Nasr A, Langer JC. Evolution of the technique in the transanal pull-through for Hirschsprung's disease: effect on outcome. J Pediatr Surg 2007; 42 (1) 36-39 , discussion 39–40
- 60 Pastor AC, Osman F, Teitelbaum DH, Caty MG, Langer JC. Development of a standardized definition for Hirschsprung's-associated enterocolitis: a Delphi analysis. J Pediatr Surg 2009; 44 (1) 251-256
- 61 Teitelbaum DH, Coran AG. Long-term results and quality of life after treatment of Hirschsprung's disease and allied disorders. In: Holschneider AM, Puri P, , eds. Hirschsprung's Disease and Allied Disorders. 3rd ed. New York: Springer; 2008: 389-397
- 62 Swenson O. How the cause and cure of Hirschsprung's disease were discovered. J Pediatr Surg 1999; 34 (10) 1580-1581
Address for correspondence
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