Journal of Pediatric Surgery 57 (2022) 69–74

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Journal of Pediatric Surgery

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Comparison of clinical outcomes after total transanal and laparoscopic

assisted endorectal pull-through in patients with rectosigmoid
Hirschsprung disease
Remi Andre Karlsen a,∗, Anders Telle Hoel a,b, Marianne Valeberg Fosby a,b, Kjetil Ertresvåg b,
Astrid Ingeborg Austrheim b, Kjetil Juul Stensrud b, Kristin Bjørnland a,b
a
Institute of Clinical Medicine, University of Oslo, Oslo, Norway
b
Department of pediatric surgery, Oslo University Hospital, Oslo, Norway

a r t i c l e i n f o a b s t r a c t

Article history: Background: Total transanal (TERPT) and laparoscopic endorectal pull-through (LERPT) are the most com-
Received 17 June 2021 mon procedures to treat rectosigmoid Hirschsprung’s disease (HD). Since few studies have compared the
Revised 10 January 2022
two methods, we aimed to assess clinical outcomes after TERPT and LERPT in this cross-sectional study.
Accepted 11 January 2022
Methods and Patients: All patients with rectosigmoid HD operated with TERPT and LERPT between 2001
and 2018 were eligible. Peri-operative data were registered from patients’ records, and bowel function
Keywords: was assessed according to the Krickenbeck classification.
Hirschsprung Results: 91/97 (94%) patients were included; 46 operated with TERPT and 45 with LERPT. Bowel function
long-term bowel function was assessed in 80 patients at median seven (4–17) years. There was no difference in functional outcome
Postoperative complications
between the procedures. Unplanned procedures under general anesthesia were frequent; 28% after TERPT
Endorectal pull-through
and 49% after LERPT (p = 0.04). 11% of TERPT and 29% of LERPT patients got botulinum toxin injections
Soiling
Botulinum toxin injection (p = 0.03). In the TERPT group, patients operated in the neonatal period had poorer outcome (78%) than
those operated later (24%) (p = 0.005). No difference in operative time, length of hospital stay, and rate
of early and late complications was found between the procedures.
Conclusion: There was no difference in long-term bowel function in patients with rectosigmoid HD op-
erated with TERPT or LERPT. More LERPT patients had an unplanned procedure under general anesthesia,
mostly due to obstructive symptoms.
Level of evidence: III.
© 2022 The Authors. Published by Elsevier Inc.
This is an open access article under the CC BY license (http://creativecommons.org/licenses/by/4.0/)

1. Introduction The literature comparing bowel function after total transanal

Endorectal pull-through (ERPT), either total transanal (TERPT) or
laparoscopic assisted (LERPT), are currently the two most common
operative procedures to treat rectosigmoid Hirschsprung’s disease
(HD) [1,2]. TERPT is the least invasive procedure with no transab-
dominal dissection and no visible scars. Furthermore, TERPT is sug-
gested to have shorter operative time and faster recovery as well
as being less expensive than LERPT [3,4]. However, there are con-
cerns that TERPT causes more anal sphincter damage because the
exposure of the anal canal is longer and more forceful when the
whole operation is performed through the anus [5–7].
Abbreviations: ERPT, Endorectal pull-through; HD, Hirschsprung’s disease; LERPT,
Laparoscopic assisted endorectal pull-through; TERPT, Total transanal endorectal
pull-through.
∗
Corresponding author at: Oslo University Hospital, Postbox 4950, Nydalen,
0424, Oslo, Norway.
E-mail addresses: remkar@ous-hf.no, remik@getmail.no (R.A. Karlsen).
and transabdominal ERPT procedures is inconclusive [4,8-11].
Transabdominal ERPT includes operations assisted with both la-
paroscopy and laparotomy, but these entities are not necessarily
identical with respect to preservation of the anal sphincters. Gen-
erally, it is easier to mobilize the rectum completely down to the
pelvic floor laparoscopically. The transanal part of the operation is
therefore shorter and technically easier. Thus, one may hypothe-
size that LERPT causes less anal sphincter damage than both TERPT
and laparotomy assisted ERPT. The importance of not damaging the
anal sphincters is well acknowledged since this may cause fecal in-
continence. Detrimental effects of fecal incontinence on psychoso-
cial health and quality of life are well documented [12]. Therefore,
it is important to study if any ERPT technique is superior with re-
spect to avoiding fecal incontinence. To the best of our knowledge,
no previous study has compared functional outcome in patients
operated with TERPT and LERPT.
HD patients and their families are not only affected by postop-
erative bowel function, but also by the number and length of hos-
pital visits, unplanned readmissions to hospital, and absence from

https://doi.org/10.1016/j.jpedsurg.2022.01.011
0022-3468/© 2022 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY license (http://creativecommons.org/licenses/by/4.0/)
70 R.A. Karlsen, A.T. Hoel, M.V. Fosby et al. / Journal of Pediatric Surgery 57 (2022) 69–74
school and work [13]. Consequently, we also sought to compare
TERPT and LERPT for other clinical outcomes than bowel function
in an attempt to identify the superiority of either TERPT or LERPT.
Thus, the main aim of this study was to compare bowel function
with specific focus on fecal incontinence after TERPT and LERPT.
Secondary aims were to compare operative time, hospital stay,
early and late complications, and unplanned postoperative proce-
dures.
2. Methods
2.1. Patients and preoperative work-up
Eligible patients with rectosigmoid aganglionosis operated with
TERPT or LERPT from 2001 to 2018 were identified by the theater
logbooks and electronic medical records. The diagnosis of HD was
verified with rectal biopsies in all patients. Contrast enemas were
used to estimate the extension of the aganglionic segment in all
patients.
2.2. Surgical management
TERPT was introduced at Oslo University Hospital in 2001.
TERPT was performed mostly as described by de la Torre et al., but
we performed a shorter mucosectomy as described by Rintala et al.
[14,15]. Briefly, the transanal mucosectomy started 0.5 to 1 cm
above the dentate line and continued for 2–3 cm, leaving a short
muscle cuff that was not split. The colonic mobilization contin-
ued well above the macroscopic transition zone, and frozen section
biopsies were taken to verify that ganglionic bowel was brought
down. From 2008, LERPT gradually replaced TERPT. The LERPT op-
erations started with obtaining a frozen section biopsy from well
above the macroscopic transition zone. Then, the sigmoid colon
and rectum were mobilized, and the dissection continued down
to the pelvic floor. The mucosectomy was performed transanally
as during TERPT. There was usually no or little need for transanal
mobilization of aganglionic rectum following the mucosectomy and
opening of the muscle cuff since this was already done laparoscop-
ically. The handsewn anastomosis was fashioned identically dur-
ing TERPT and LERPT. The anastomosis was calibrated two to three
weeks postoperatively and then according to surgeons’ preference,
but at least every three weeks for the first two months.
2.3. Definitions
Complications occurring the first 30 postoperative days were
graded according to the Clavien-Dindo classification system [16].
Bowel function was assessed with the Krickenbeck classification in
patients older than four years who did not have a stoma or used
bowel management [17]. Bowel management implied regular use
of antegrade or rectal enemas. Perineal excoriations were wounds
problematic to handle as noted by nurses and/or doctors in the
patients’ medical records. Stricture was defined as the need for di-
latation/calibration of the anastomosis beyond the third postoper-
ative month and/or dilatation requiring general anesthesia at any
time. Soiling was defined as involuntary leaking of small amounts
of stool, requiring change of underwear or diapers. Poor outcome
was defined as daily soiling, use of bowel management or having
a stoma. Intended TERPT procedures converted to laparoscopy or
laparotomy were included in the TERPT group. Any post-ERPT pro-
cedure under general anesthesia that might be related to the ERPT,
excluding stoma-related operations in a multi-staged ERPT, was de-
fined as an unplanned procedure.
2.4. Data collection
Patient demographics, operative details, complications, and un-
planned procedures were recorded from medical records. Opera-
tion time was not recorded if concomitant procedures were per-
formed. Patients and/or parents answered the Krickenbeck ques-
tionnaire and a self-designed questionnaire covering use of bowel
medication, bowel management, and if they had a stoma. The
questionnaires were filled out during out-patient visits or sent by
mail if no out-patient visit was scheduled during 2019. The sur-
geon(s) in charge of the treatment was not present when the ques-
tionnaires were filled out. Bowel function, according to the Krick-
enbeck classification, was only assessed in patients over four years
who did not use bowel management and did not have a stoma. Pa-
tients and/or parents with language problems making reliable as-
sessment of bowel function impossible did not fill out any ques-
tionnaires. To compare bowel function at most similar ages in
the two groups, bowel function assessments from TERPT patients
recorded in 2008 were used when available [11].
Guidelines for Strengthening the Reporting of Observational
Studies in Epidemiology (STROBE) were applied [18].
2.5. Statistics
Categorical variables were analyzed with chi square test or
Fisher’s exact test as appropriate. Continuous variables were pre-
sented as median (min-max) and analyzed with t-test or Mann-
Whitney U test as appropriate. Statistical significance was set at
p<0.05. IBM SPSS software for Windows version 25 (Armonk, NY:
IBM Corp.) was used.
2.6. Ethics
The study was approved by the institutional review board
(2017/4913). Written informed consent was obtained.
3. Results
3.1. Patients
Of 97 eligible patients, 91 (94%) were included in the study; 46
in the TERPT group and 45 in the LERPT group (p = 1) (Fig. 1).
Questionnaires were sent to 16 patients, and 10 responded. Except
for age at diagnosis and operation, patient demographics were not
different between the groups (Table 1). Ten (11%) patients had vari-
ous syndromes; Down syndrome (7), Arnold-Chiaris syndrome (1),
Goldberg-Shprintzen syndrome (1), or a mosaicism condition (1).
Other comorbidities included heart disease (4), diaphragmatic her-
nia (2), hearing loss (2), esophageal atresia (1), undescended testis
(1), hydronephrosis (1), hypospadia (1), trigonocephaly (1), spina
bifida (1), and clubfoot (1).
3.2. Pre- and perioperative stomas
A diverting stoma prior to the ERPT was created in three (7%)
TERPT patients and 11 (24%) LERPT patients (p = 0.02). The three
TERPT patients got a preoperative stoma because rectal stimulation
and irrigation did not ensure sufficient bowel emptying, and pri-
mary ERPT was considered unsafe because of Hirschsprung associ-
ated enterocolitis, poor general condition or low weight. Reasons
for creating a preoperative stoma in the LERPT group were hugely
dilated colon (9) and bowel obstruction prior to diagnosis of HD
(2). Furthermore, four LERPT patients got a diverting stoma con-
comitantly with the ERPT due to massive colonic dilatation. One
patient in each group had their preoperative stoma reversed dur-
ing the ERPT. All stomas established either before or concomitantly
 R.A. Karlsen, A.T. Hoel, M.V. Fosby et al. / Journal of Pediatric Surgery 57 (2022) 69–74 71

Fig. 1. Flow diagram of inclusion and data collection.

Table 1
Demographics of patients operated with total transanal (TERPT) or laparoscopic assisted
endorectal pull-through (LERPT). Continuous variables are presented as median (min-max).

TERPT LERPT p
(n = 46) (n = 45)

Boys 37 (80%) 36 (80%) 0.96

Syndromic patients 6 (13%) 4 (9%) 0.74
Non-syndromic patients with comorbidities 3 (7%) 7 (16%) 0.2
Age at diagnosis (months) 0.6 (0.1–117) 4.0 (0.1–95) 0.03
Age at operation (months) 1.9 (0.4–133) 5.5 (0.6–98) 0.02
Age at bowel function assessment (years)1 7.75 (4–17) 6.6 (4–14) 0.1
1
Bowel function was assessed in 80 patients >4 years of age without language prob-
lems.
72 R.A. Karlsen, A.T. Hoel, M.V. Fosby et al. / Journal of Pediatric Surgery 57 (2022) 69–74

Table 2 while the patient had a stoma. Furthermore, nine stoma revisions
Complications during the first 30 postoperative days after total transanal
were performed. The two patients with anastomotic leakage and
(TERPT) or laparoscopic assisted endorectal pull-through (LERPT). Some
patients had more than one complication. Stoma related complications two patients with obstructive symptoms had their stomas taken
are not included. Complications are graded according to the Clavien down. One of the two LERPT patients with anastomotic leakage
Dindo classification system. was later diagnosed with twisted colon and reoperated with a
TERPT LERPT p colocolostomy.
(n = 46) (n = 45)

Patients with complications 16 (35%) 18 (40%) 0.61 3.5. Long-term bowel function
Grade I complications 8 (17%) 6 (13%) 0.59
Perineal excoriations 8/441 (18%) 5/311 (16%)
Gastroenteritis 0 1 (2%)
Eighty patients filled out the self-designed questionnaire
Grade II complications 9 (20%) 12 (27%) 0.42 (Fig. 1); 70 (88%) during out-patient visits and 10 (12%) by
Wound infection2 2 (4%) 0 mail. Sixty-one patients filled out the Krickenbeck questionnaire
Infection, unknown focus 4 (9%) 6 (13%) (Table 4).
Obstructive symtoms3 3 (7%) 6 (13%)
No soiling, no constipation, no use of bowel management and
Grade IIIa complications 0 0
Grade IIIb complications 1 (2%) 4 (9%) 0.2 no stoma, were reported by 20/46 (43%) and 13/34 (38%) of TERPT
HAEC4 0 1 (2%) and LERPT patients, respectively (p = 0.64). Sixteen of 46 (35%)
Infection, unknown focus5 1 (2%) 0 TERPT patients and 12/34 (35%) LERPT patients had poor bowel
Anastomotic stricture 0 1 (2%)
function (p = 0.96). Any report of soiling, either in the Krickenbeck
Anastomotic leakage6 0 2 (4%)
questionnaire or as a reason for bowel management, was reported
1
Excluding patients with a postoperative stoma. 2 Surgical site infec- by 12/46 (26%) TERPT patients and 11/34 (32%) of LERPT patients
tion after conversion from TERPT to laparotomy. 3 Antibiotics and rec-
(p = 0.47). When syndromic patients were excluded from the anal-
tal irrigations were given because of obstructive symptoms to pre-
vent Hirschsprung associated enterocolitis (HAEC). 4 Bowel irrigations
yses, postoperative bowel function results remained similar for the
were done under general anesthesia. 5 Central venous catheter insertion. two operative techniques (results not shown).
6
Patients got diverting stoma and drainage of abscess. Nine of 10 patients operated in the neonatal period underwent
TERPT. Of these nine TERPT patients, seven (78%) reported poor
outcome at follow-up compared to 9/37 (24%) TERPT patients op-
with the ERPT, were reversed at median 1.7 (1.0–2.6) and 1.8 (0.8–
erated beyond the neonatal period (p = 0.005).
7.8) months after TERPT and LERPT, respectively.

3.3. Surgical details and postoperative complications 4. Discussion

TERPT was performed by nine surgeons, whereas LERPT was
performed by five surgeons, of whom four had experience with
TERPT. Median operative time for TERPT was 158 (84–318) min
and 176 (95–318) min for LERPT (p = 0.34). Hospital stay was me-
dian six days after both TERPT (3–18) and LERPT (3–32) (p = 0.50).
Two intended TERPT procedures were converted to laparotomy or
laparoscopy, because of multiple adhesions and difficult colonic
mobilization.
Early complications were common after both TERPT and LERPT
(Table 2). Excluding stoma related complications, 16 (35%) TERPT
patients experienced a total of 18 complications, and 18 LERPT pa-
tients (40%) experienced a total of 22 complications. There was
no significant difference in overall complication rates between
the procedures. Clavien-Dindo grade IIIb complications were reg-
istered in 2% of TERPT patients compared to 9% of LERPT patients
(p = 0.20).
Anastomotic stricture occurred in 16 (35%) TERPT patients and
nine (20%) LERPT patients (p = 0.11). The strictures were treated
with anal dilatations for median five (3–12) months after TERPT
and four (3–12) months after LERPT (p = 0.53). All dilatations were
done during the first two postoperative years, and no patients re-
quired reoperation due to stricture.
3.4. Unplanned procedures
Unplanned procedures were common in both groups, with bo-
tulinum toxin injections and dilatations being the most frequent
(Table 3). Botulinum toxin injections were given to patients with
obstructive symptoms that did not respond to laxatives, and was
introduced at our center in 2005. Botulinum toxin injections were
significantly more often administered in LERPT patients (p = 0.03).
Three TERPT and five LERPT patients got a stoma after the ERPT
(p = 0.49) due to anastomotic leakage (2 LERPT) or severe ob-
structive symptoms (3 TERPT, 3 LERPT). One TERPT patient with
obstructive symptoms developed a rectal prolapse that resolved
The main finding from this study is that operative approach, to-
tal transanal or laparoscopy assisted ERPT, does not significantly
affect long-term bowel function in HD patients with rectosigmoid
aganglionosis. Thus, our hypothesis that TERPT causes more dam-
age to the anal sphincters and thereby more fecal incontinence
than LERPT, was not supported. Our data are in line with a re-
cent review comparing results after transanal and transabdominal
ERPT procedures in 476 patients [4]. In this review, however, most
transabdominal operations were done by laparotomy. It seems that
other factors than the operative approach are more important for
postoperative bowel function. These include the level of the anas-
tomosis above the dentate line, motility of the remnant ganglionic
colon, immunologic and microbiological aspects of HD, and age at
operation. Data have shown that damage to the anal canal and a
too low anastomosis cause fecal incontinence [19,20]. Since dys-
motility in the upper gastrointestinal tract has been demonstrated
in HD patients, it is not surprising that dysmotility also has been
found in the remnant ganglionic colon [21–23]. A recent study
found increased expression of protease-activated receptors in HD
patients’ colon that could be linked to inflammation, permeabil-
ity and gut motility [24]. Lastly, novel animal studies have identi-
fied a microbiota-driven crosstalk between macrophages and en-
teric neurons that controls gastrointestinal motility, further sug-
gesting that many factors besides operative procedure affect post-
operative bowel function in HD patients [25].
An interesting finding in this study is that patients operated
with TERPT in the neonatal period had a higher risk of poor bowel
function than those operated later. TERPT is considered a safe pro-
cedure in neonates. However, some studies have found more early
complications and more fecal incontinence after neonatal TERPT
[26,27]. Plausible explanations are iatrogenic injury to the anal
canal and the anal sphincters. Current ERNICA guidelines suggest
doing ERPT at 2–3 months of age if the child is growing well and
the bowel is sufficiently decompressed [19,28,29]. Similarly, a re-
cent multicenter, retrospective study found that delayed primary
 R.A. Karlsen, A.T. Hoel, M.V. Fosby et al. / Journal of Pediatric Surgery 57 (2022) 69–74 73

Table 3
Unplanned procedures under general anesthesia after total transanal (TERPT) or laparoscopic
assisted endorectal pull-through (LERPT). Some patients had more than one unplanned pro-
cedure.

TERPT LERPT
(n = 46) (n = 45) p

Patients with any unplanned procedure 13 (28%) 22 (49%) 0.04

Patients getting botulinum toxin injections 5 (11%) 13 (29%) 0.03
Patients undergoing anal dilatations 7 (15%) 7 (16%) 0.96
Total number of unplanned procedures 74 155
Anal dilatations (total number) 48 70
Number of dilatations/patient, median (min-max) 3 (1–21) 7 (2–38)
Botulinum toxin injections (total number) 14 65
Number of injections/patient, median(min-max) 3 (1–5) 4 (1–16)
Stoma creations, revisions and reversals1 7 14
Appendicostomy 5 5
Reoperation (for twisted colon) 0 1
1
Stoma creations or reversals as part of a multi-stage endorectal pull-through are not
included.

Table 4
Long-term outcome in Hirschsprung patients > 4 years operated with either total transanal endorectal
pull-through (TERPT) or laparoscopic assisted endorectal pull-through (LERPT). Bowel function, described
according to the Krickenbeck classification, was not reported in patients who used regular bowel man-
agement (regular use of enemas) or had a stoma.

TERPT LERPT
N = 46 N = 34 p

Patient answering the Krickenbeck questionnaire N = 35 (76%) N = 26 (76%)

Voluntary bowel movements1
Yes 29 (63%) 20 (59%) 0.56
No 6 (13%) 6 (18%)
Soiling
No 25 (54%) 19 (56%) 0.29
Grade 1: Occasionally (once or twice per week) 5 (11%) 3 (9%)
Grade 2: Every day, no social problem 3 (7%) 0
Grade 3: Constant, social problem 2 (4%) 4 (12%)
Constipation
No 29 (63%) 20 (59%) 0.56
Grade 1: Manageable with diet 0 0
Grade 2: Requires laxatives 5 (11%) 4 (12%)
Grade 3: Resistant to diet and laxatives 1 (2%) 2 (6%)
Patients using bowel management or having a stoma N = 11 (24%) N = 8 (24%)
Bowel management 10 (22%) 6 (18%) 0.65
Appendicostomy 5 (11%) 5 (15%)
Retrograde enemas 5 (11%) 1 (3%)
Stoma2 1 (2%) 2 (6%) 0.57
1
One TERPT patient had constipation and soiling, and was scored for both. Feeling of urge, capacity to
verbalize, and ability to hold the bowel movement.

ERPT was at least as good as a neonatal operation [30]. Thus, our
results support these guidelines and findings.
The high number of unplanned procedures in the LERPT group,
mostly botulinum toxin injections due to obstructive symptoms, is
worrying. There are no studies that specifically compare frequency
of botulinum toxin injections after TERPT and LERPT. Several re-
cent non-comparative studies find that LERPT patients get bo-
tulinum toxin injections more often than TERPT patients [5,31,32].
This may indicate a true difference between the surgical proce-
dures. However, it is possible that treatment of obstructive symp-
toms has become more aggressive in recent years when LERPT has
gained popularity. It could also be that increased focus on preser-
vation of the anal canal has made surgeons fashioning the anas-
tomosis higher above the dentate line and thereby cause more
obstruction.
Few patients reported constipation, and there was no difference
between TERPT and LERPT patients. The constipation rate in this
patient population is within the range of other reports (0–35%) [4].
Interestingly, as noted in one previous study, none of those who re-
ported constipation could manage with dietary precautions alone
[33]. Since diet does not seem to effectively reduce constipation
in HD patients, health care providers should avoid too much fo-
cus on strict anti-constipation diets. We have previously reported
that adult HD patients have a great interest in diet and would
like to get advice from a dietician [34]. Since effects of diet are
scarcely studied in HD patients, future studies should aim at ob-
taining more knowledge about the influence of diet on bowel func-
tion in HD patients.
The rate of major complications after both LERTP and TERPT is
around 10% [35]. How major complications are defined, varies be-
tween studies. The rate of Clavien-Dindo grade IIIb complications
in this study is in line with previous literature. Due to the low
number of patients, we cannot decide which procedure has the
lowest complication rate. Interestingly, the only two anastomotic
leakages were in the LERPT group. Intraabdominal overview to
avoid twisting of the pulled-through colon has been advocated as
one of the advantages of LERPT. Interestingly, the only patient ex-
periencing this complication underwent LERPT demonstrating that
even with laparoscopy one has to be very careful to avoid twisting.
Another advantage of LERPT is the possibility to take a biopsy at
the start of the operation, as long segment HD cannot be ruled out
by contrast enema alone. Since 2005, patients undergoing TERPT
74 R.A. Karlsen, A.T. Hoel, M.V. Fosby et al. / Journal of Pediatric Surgery 57 (2022) 69–74
in our institution had a transumbilical biopsy at the start of the
operation [36].
The strengths of this study are the high response rate, the use
of an established bowel function questionnaire, and that the clini-
cal evaluation was done without the performing surgeon present. A
relatively small patient population, the retrospective study design,
and patients operated in different time periods by partly different
surgeons are the main limitations. The different time periods for
the operations exclude selection bias due to surgeons’ preference,
but there could be a learning curve advantage as well as minor
changes in follow up routines during the timeframe of the study.
Furthermore, there was a small age difference between the groups
at assessment of bowel function. Based on previous studies, we do
not think this has significantly influenced the results [37].
In conclusion, whether ERPT was performed total transanally
or laparoscopy assisted did not affect long-term bowel function.
Unplanned procedures, particularly due to obstructive symptoms,
were more common in the LERPT group.
Funding
This study was partly funded by the Dam Foundation and the
University of Oslo.
Conflicts of interest statement
The authors declare no conflicts of interest.
Supplementary materials
Supplementary material associated with this article can be
found, in the online version, at doi:10.1016/j.jpedsurg.2022.01.011.
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