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Article history: Objective: To compare treatment outcomes in children with Hirschsprung's disease who underwent treatment
Received 17 July 2017 using the Duhamel or TERPT surgical procedures.
Received in revised form 28 September 2017 Methods: Medline, Cochrane, EMBASE, and Google Scholar databases were searched through December 26, 2016.
Accepted 10 October 2017 Search strings included Hirschsprung's disease, fecal incontinence, transanal endorectal pull-through, and Duhamel
operation. Randomized controlled studies (RCTs) and retrospective studies that compared the treatment of
Key words:
Hirschsprung's disease in with TERPT or Duhamel surgical procedures in neonates, infants, or children were included.
Duhamel operation
Transanal endorectal pull-through procedure
Results: The study included six studies with a total of 280 patients. The meta-analysis indicated that the Duhamel
TERPT and TERPT interventions were similar with respect to rate of postoperative fecal incontinence (OR = 0.85, 95%
Hirschsprung disease CI = 0.37 to 1.92, P = 0.692) and operation time (difference in means = 46.68 min, 95% CI = − 26.96 to
Fecal incontinence 114.31, P = 0.226). The Duhamel procedure was associated with longer postoperative hospital stay (Difference
in means = 3.14 days, 95% CI = 1.46 to 4.82, P b .001) and a lower rate of enterocolitis (OR = 0.21, 95% = 0.07 to
0.68, P = 0.009) compared with the TERPT procedure.
Conclusions: The study found that Duhamel and TERPT procedures showed similar benefit in treating
Hirschsprung's disease, although differences exist with respect to length of postoperative hospital stay and the
incidence of enterocolitis.
The type of study: Meta-analysis.
Level of evidence: Level II.
© 2017 Elsevier Inc. All rights reserved.
Hirschsprung's disease is a congenital disease that is estimated to anus [8]. The Soave procedure involves removal of the rectal mucosa
occur in one out of 5000 births [1–3]. A variety of definitive surgical pro- while retaining the muscular cuff and a ganglionic segment of colon is
cedures are used to treat Hirschsprung's disease, with most cases having anastomosed to the mucosa of the anal canal [2].
pull-through procedures. The purpose of a pull-through procedure is to The transanal endorectal pull-through (TERPT) was originally a
remove the aganglionic colon, bring normally innervated bowel to the Soave-like transanal submucosal dissection with an endorectal pull-
anus and preserve anal sphincter function [4]. through leaving an aganglionic rectal muscular cuff [7,9]. The procedure
Several procedures have been used to treat the disease [5–7]. One has been modified to a transanal Swenson-like operation in which dis-
commonly used technique is the Duhamel retrorectal pull-through pro- section in the submucosal plain is not required; instead a straight resec-
cedure. The Duhamel technique involves a retrorectal transanal pull- tion of the full-thickness of the colon above the dentate line is used
through method and does not require resection of the rectum [2]. The [4,10]. Laparoscopy can be used for localization of the transition zone
posterior wall of the rectum and the anterior wall of the pulled- and for mobilization of the aganglionic distal sigmoid colon [10].
through colon are opposed by a crushing clamp resulting in a wide anas- Over the past several years, some centers of pediatric surgery have
tomosis [2]. Therefore, a section of aganglionic rectum is left connected transitioned from performing the Duhamel procedure to performing
to a segment of ganglionic colon (side-to-side) as a pouch reservoir [2]. the TERPT procedure for most cases [10]. Both the TERPT technique
The Swenson, Rehbein and Soave procedures are also used to treat and the Duhamel technique can be performed via laparoscopic surgery,
Hirschsprung's disease. The Swenson procedure involves an abdominal which results in less trauma, lower amounts of blood loss, less intraper-
incision and extra mucosal biopsies are taken along the antimesenteric itoneal contamination, and less intestinal adhesion [10–12]. However, it
border and assessed by frozen section to determine the level of gangli- is unclear if one of these two techniques yields significantly better
onated bowel. The operation involves removing the aganglionic rectum, disease-specific and general outcomes. It is also not clear which method
pulling the healthy ganglionated colon through, and connecting it to the results in the least amount of complications, shorter hospital stays, and
shorter operation time, all of which can impact outcomes and medical
costs. The aim of this systematic review and meta-analysis was to eval-
⁎ Corresponding author. Tel: +86 27 85726005. uate the relative benefits of TERPT and Duhamel procedure in treating
E-mail address: maoyz68@126.com (Y. Mao). Hirschsprung's disease.
https://doi.org/10.1016/j.jpedsurg.2017.10.047
0022-3468/© 2017 Elsevier Inc. All rights reserved.
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Y. Mao et al. / Journal of Pediatric Surgery 53 (2018) 1710–1715 1711
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1712 Y. Mao et al. / Journal of Pediatric Surgery 53 (2018) 1710–1715
were included in the meta-analysis. A random-effects model of analysis 2.4. Postoperation hospital stay (postoperation hospital stay, days)
was used as a large degree of heterogeneity in the data was seen (Q sta-
tistic =16.59; I2 = 87.95%; P b .001). The overall pooled analysis found Postoperation length of hospital stay was reported in the studies of
that the operation time was similar between the two surgical proce- Sosnowska et al. (2016), Gunnarsdóttir et al. (2010), and Tannuri et al.
dures (difference in means = 46.68 min., 95%CI = −26.96 to 114.31, (2009). A fixed-effects model of analysis was used as no heterogeneity
P = 0.226) (Fig. 3). was seen in the data (Q statistic =1.35; I2 = 0%; P = 0.509). The overall
Table 1
Basic characteristics of studies for meta-analysis.
Study number 1st AU (Year) Study design Group Number of patients Age at diagnosis Age at operation Males, Length of follow up Quality assessment
(mean, month) (mean, month) (%) (mean, month) (NOS)
Abbreviations: 1st AU, first author; LTEPT, laparoscopic-assisted transanal endorectal pull-through; mo, month; n/a, not available; NOS, Newcastle–Ottawa scale; TERPT, transanal
endorectal pull-through techniques.
a
Median.
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Y. Mao et al. / Journal of Pediatric Surgery 53 (2018) 1710–1715 1713
Table 2
Summary of clinical outcomes and complications.
Study 1st AU (Year) Group Fecal Fecal Constipation, Operation time, Postoperation Enterocolitis,
number incontinence, Continence, n (%) (min) hospital stay, (day) (n)
n (%) n (%)
Obstruction: 10%
Sosnowska Duhamel 1 (3%) n/a 240 (120–360)a 17 (9–60)a
1 Stool incontinence: 3% 2
(2016)
TERPT 0% 0% n/a 230 (120–270)a 17 (7–40)a
good and normal
Duhamel n/a n/a
Tannuri continence: 70%
2
(2017) good and normal
TERPT n/a n/a
continence: 47.6%
Giuliani Duhamel n/a n/a 2 (6.1%) 257 7 1
3
(2011) TERPT n/a n/a 0% 195 4 0
Gunnarsdóttir Duhamel 3 (18%) n/a 10 (59%) 154 ± 35 6.9 ± 3.8 2
4
(2010) TERPT 1 (8%) n/a 3 (27%) 146 ± 25 4.4 ± 1.5 2
Complete continence:
17 (58.6%)
Duhamel 2 (6.9%) 6 (20.7%) 232 ± 82.7 8.4 ± 6.3 1
Partial continence:
Tannuri 10 (34.5%)
5
(2009) Complete continence:
17 (70.8%)
TERPT 2 (8.3%) 0% 120 ± 29.2 4.3 ± 3.69 7
Partial continence:
5 (20.8%)
satisfactory: 16 (48%)
Minford Duhamel 17 (52%) (Functional continence n/a n/a n/a 1
6
(2004) score)
TERPT 20 (59%) satisfactory: 14 (41%) n/a n/a n/a 13
Abbreviations: 1st AU, first author; mo, months; h, hours; min., minutes; n/a, not available.
a
Mean (range).
analysis indicated that patients with Duhamel intervention had longer did not markedly differ with the omission of any one study, indicating
postoperation hospital stay compared with patients who were treated that the meta-analyses were robust and that the data were not overly
with TERPT intervention (Difference in means = 3.14 days, 95% CI influenced by any study.
=1.46 to 4.82, P b .001) (Fig. 4).
2.7. Quality assessment
2.5. Enterocolitis rate
Quality of the included studies was assessed using the Newcastle–
The studies of Giuliani et al. (2011), Gunnarsdóttir et al. (2010), Ottawa scale. Four of the included studies had score ≥ 6, with the
Tannuri et al. (2009), and Minford et al. (2004) reported data for the in- other two studies scoring 4 and 5. Major limitation of the studies was
cidence of enterocolitis. A fixed-effects model of analysis was used as the lack of community controls, which is related to study characteristics
low level of heterogeneity was observed (Q statistic = 3.75; I 2 = and design. Overall the included studies were of acceptable quality.
20.07%; P = 0.289). The overall analysis revealed that patients treated
by the Duhamel procedure had lower rate of enterocolitis than those 3. Discussion
treated using the TERPT technique (OR = 0.21, 95% CI = 0.07 to 0.68,
P = 0.009) (Fig. 5). The objective of the current study was to compare treatment out-
comes in children with Hirschsprung's disease who underwent treat-
2.6. Sensitivity analysis ment using the Duhamel or TERPT surgical procedures. Six studies
were included. The meta-analysis indicated that the Duhamel and
Sensitivity analysis was performed on each outcome using the leave- TERPT interventions were similar with respect to rate of postoperative
one-out approach in which a pooled analysis was performed after each fecal incontinence and operation time. The Duhamel procedure was as-
study was left out in turn (Supplemental Fig. 1A, B, C, and D). For the sociated with longer postoperation hospital stay and lower rate of en-
four outcomes, the direction and magnitude of the combined estimates terocolitis compared with the TERPT procedure. To our knowledge,
Fig. 2. Forest plot for comparing fecal incontinence rate between patients treated with Duhamel or TERPT procedures. Abbreviations: CI, confidence interval; Lower limit, lower bound of
the 95% CI; Upper limit, upper bound of the 95% CI.
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1714 Y. Mao et al. / Journal of Pediatric Surgery 53 (2018) 1710–1715
Fig. 3. Forest plot comparing operation time between patients treated using Duhamel or TERPT procedures. Abbreviations: CI, confidence interval; Lower limit, lower bound of the 95% CI;
Upper limit, upper bound of the 95% CI.
this is the first systematic review and meta-analysis that specifically procedure. These findings suggest that infants should be followed close-
compared treatment outcomes following Duhamel and TERPT surgery ly for enterocolitis, particularly those who were treated using the TERPT
in patients with Hirschsprung's disease. surgical approach. Follow-up should be at least for two years as most
A prior systematic review and meta-analysis performed by Chen et al. postoperative cases of enterocolitis occur within two years of ileoanal
(2013) compared clinical outcomes of conventional transabdominal ap- pull-through anastomosis [21].
proach (ie, Duhamel, Swenson, Soave, and Rehbein procedures) with The current study has several limitations. All the included studies
the TERPT procedure [20]. The study of Chen et al. included 93 studies. were retrospective or prospective studies and the patient population
They found that TERPT was associated with shorter operative time was small; no RCTs were included. In addition, in several of the included
and hospital stay, as well as, less postoperative incontinence/soiling studies, the age at the time of operation for the TERPT group was youn-
and constipation (P values ≤0.010) compared with transabdominal ap- ger than that of the Duhamel group, which may have led to potential
proaches. They found no difference between approaches in postopera- bias and possibly confounded the findings. It was not possible to assess
tive enterocolitis. The difference between the findings of Chen et al. the incidence of constipation following surgery, which reflects a limita-
and our study may reflect the fact that we only compared TERPT and tion of the statistical analysis which cannot assess an incidence of 0%
Duhamel procedures while their analysis pooled data across studies (see Giuliani et al. [2011] and Tannuri et al. [2009]) and the low
that evaluated different types of transabdominal approaches for power seen in the other two papers. Additional trials are necessary to
treating Hirschsprung's disease. In addition, most of the studies includ- further compare these two surgical methods for treating Hirschsprung's
ed in their meta-analysis compared TERPT with the Soave procedure. disease.
Chen et al. did not perform subgroup analysis to evaluate Duhamel In summary, the findings of this study indicated that Duhamel
and TERPT techniques. In addition, Chen et al. included studies per- and TERPT surgical approaches for treating infants and children
formed in infants, children and adults, while in the current study, we fo- with Hirschsprung's disease were similar with respect to postopera-
cused on those performed in infants and children. Similar to our study, tive fecal incontinence and operation time. However, TERPT proce-
Chen et al. observed a large degree of heterogeneity among the studies dure was associated with shorter length of postoperation hospital
in operative time, which may reflect variation in the skill of the surgeon stay, while the Duhamel technique resulted in a lower rate of
[20]. The current study did not evaluate the incidence of postoperative enterocolitis. The impact of these differences on long-term patient
constipation. outcomes and medical costs is unclear. Further studies are warrant-
Most patients treated for Hirschsprung's disease do not experience ed to further evaluate the surgical options used for treating this
complications; however up to about 10% may have constipation and disease.
b 1% have fecal incontinence [8]. Enterocolitis and colonic rupture are Supplementary data to this article can be found online at https://doi.org/
the most serious disease-related complications and are major causes 10.1016/j.jpedsurg.2017.10.047.
of mortality associated with this disease. Enterocolitis occurs in up to
50% of infants with Hirschsprung's disease and is commonly because
of intestinal obstruction and residual aganglionic bowel [21]. In the
studies included in the current analysis, in general, the incidence of en- Declaration of funding
terocolitis was low; however, TERPT technique was associated with a
greater incidence of enterocolitis compared with the Duhamel None.
Fig. 4. Forest plot comparing length of postoperative hospital stay between patients treated with Duhamel or TERPT procedures. Abbreviations: CI, confidence interval; Lower limit, lower
bound of the 95% CI; Upper limit, upper bound of the 95% CI.
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Y. Mao et al. / Journal of Pediatric Surgery 53 (2018) 1710–1715 1715
Fig. 5. Forest plot comparing rate of enterocolitis between patients treated with Duhamel or TERPT procedures. Abbreviations: CI, confidence interval; Lower limit, lower bound of the 95%
CI; Upper limit, upper bound of the 95% CI.
Declaration of financial/other relationships [10] Arts E, Botden SM, Lacher M, et al. Duhamel versus transanal endorectal pull through
(TERPT) for the surgical treatment of Hirschsprung's disease. Tech Coloproctol 2016;
20:677–82.
None. [11] Zhang S, Li J, Wu Y, et al. Comparison of laparoscopic-assisted operations and lapa-
rotomy operations for the treatment of Hirschsprung disease: evidence from a
meta-analysis. Medicine (Baltimore) 2015;94:e1632.
Acknowledgments [12] Gosemann JH, Friedmacher F, Ure B, et al. Open versus transanal pull-through for
Hirschsprung disease: a systematic review of long-term outcome. Eur J Pediatr
Surg 2013;23:94–102.
None. [13] Lee J, Kim HS, Shin SH, et al. Efficacy and safety of fluconazole prophylaxis in ex-
tremely low birth weight infants: multicenter pre-post cohort study. BMC Pediatr
2016;16:67.
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