Journal of Pediatric Surgery 49 (2014) 560–563

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Long term outcomes of laparoscopic-assisted anorectoplasty: A comparison study with posterior sagittal anorectoplasty An-Xiao Ming a, Long Li a,⁎, Mei Diao a, Hai-Bin Wang a, Yao Liu a, Mao Ye a, Wei Cheng b,⁎⁎ a

Department of Pediatric Surgery, Capital Institute of Pediatrics, Beijing 100020, P. R. China Department of Paediatric Surgery, Monash Children’s, Monash Medical Center, Southern Health, Department of Paediatrics and Department of Surgery, Southern Medical School, Faculty of Medicine, Nursing and Health Sciences, Monash University, Clayton, Victoria 3168, Australia b

a r t i c l e

i n f o

Article history: Received 14 October 2012 Received in revised form 23 October 2013 Accepted 18 November 2013 Key words: Anorectal malformation Recto-bladderneck and recto-prostatic fistula Laparoscope Functional evaluation

a b s t r a c t Purpose: The aim of this study is to compare the long term outcomes between laparoscopic-assisted anorectoplasty (LAARP) and posterior sagittal anorectoplasty (PSARP) for children with rectobladderneck and rectoprostatic fistula anorectal malformations (ARM). Methods: Thirty-two ARM children with rectobladderneck and rectoprostatic fistula who underwent LAARP between October 2001 and March 2012 were reviewed. The outcomes were compared with those of 34 ARM children who underwent PSARP between August 1992 and September 2001. The sacral ratio (SR), age at operation, operative time, postoperative hospital stay and complications were evaluated. Bowel functions were assessed using the Krickenbeck classification. Results: The mean operative time of the LAARP was significantly shorter than that of PSARP group (1.62 ± 0.40 vs 2.13 ± 0.30 h). The postoperative hospital stay was significantly shorter in the LAARP group (5.8 ± 0.65 vs 8.4 ± 0.67 h). The wound infections (11.8% vs 0%) and recurrent fistula (11.8% vs 0%) were more common in PSARP patients. The overall morbidity rate of PSARP group was significantly higher than that of the LAARP group (35.3% vs 12.5%, p b 0.05). However, 7.5% of the LAARP patients developed rectal prolapse. Twenty-four of 32 patients were followed up for more than 3 years in LAARP group. The median follow up period was 7.5 years (range 4–11) in LAARP patients and 15.5 years (range 11–20) in PSARP patients. The rates of voluntary bowel movement, soiling (grade 1, 2 & 3) were similar in both groups. More patients from PSARP group developed grade 2 or 3 constipation (22.5% vs 0%, P b 0.01). Conclusions: Compared to PSARP, LAARP is a less invasive procedure. The long term functional outcomes after LAARP were equivalent if not better than those of PSARP. © 2014 Elsevier Inc. All rights reserved.

Anorectal malformation with recto-bladderneck and recto-prostatic fistula represents a challenge for pediatric surgeons. Posterior sagittal anorectoplasty (PSARP) is commonly employed as a treatment for high anorectal malformations. However, laparoscopic assisted anorectoplasty (LAARP)is becoming increasingly adopted in the management of patients with high/intermediate ARM [1,2]. The functional benefit of LAARP remains unclear. The purpose of this study is to assess the long term outcomes of laparoscopic-assisted anorectoplasty (LAARP) for children with rectobladderneck and rectoprostatic fistula anorectal malformations.

⁎ Correspondence to: L. Li, Department of Pediatric Surgery Capital Institute of Pediatrics, Beijing, P. R. China. Tel.: +86 10 8569 5669. ⁎⁎ Correspondence to: W. Cheng, Department of Paediatric Surgery, Monash Children’s, Southern Health, Department of Paediatrics and Department of Surgery, Southern Medical School, Faculty of Medicine, Nursing and Health Sciences, Monash University, Victoria, Australia. Tel.: +61 3 9594 5674; fax: +61 3 9594 6495. E-mail addresses: [email protected] (L. Li), [email protected] (W. Cheng). 0022-3468/$ – see front matter © 2014 Elsevier Inc. All rights reserved. http://dx.doi.org/10.1016/j.jpedsurg.2013.11.060

1. Materials and method 1.1. Patient demographics Thirty-two ARM children with recto-bladderneck and rectoprostatic fistula who successfully underwent LAARP between October 2001 and March 2012 were reviewed. The outcomes were compared with those of 34 children who underwent PSARP between August 1992 and September 2001 in our institution. Operations in the both groups were performed by the same surgeon. The median follow up periods for LAARP and PSARP groups were 6.3 (range: 0.5–11) years and 15.5 (range: 11–20) years, respectively. Anorectal anomalies were categorized according to the Krickenbeck classification [3]. Colostograms were conducted to identify the type of malformations (Fig. 1). In the LAARP group, there were 18 recto-prostatic fistulae and 14 recto-bladderneck fistulae. In the PSARP group, there were 17 recto-prostatic fistulae and 17 recto-bladderneck fistulae. Patients were reviewed retrospectively. Sacral ratio (SR), age at operation, operative time, postoperative hospital stay and complications were compared.

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Fig. 2. MRI shows a centrally placed rectum.

2. Results

Fig. 1. Colostogram of recto-prostatic fistulae.

1.2. Complications The post-operative complications of the two groups were compared. We looked into wound infections, rectal retraction, recurrent fistula, urethral diverticulum, anal stenosis and rectal prolapse. Urethral diverticulum was evaluated by retrograde cystography and MRI. Imaging of the position of the colon within the muscle complex was also assessed by MRI. Abnormality was defined as an incomplete muscle ring on any side, inequality of the muscle along the circumference, or a misplaced rectum outside the muscle along any length of the tunnel [4]. 1.3. Functional results Functional results of patients older than 3 years were analyzed. Bowel functions including presence of voluntary bowel movements, soiling and constipation, were assessed according to the Krickenbeck classification [3]. We considered it a good outcome when the patients were free from soiling or suffer from grade 1 soiling only and a poor outcome for those presenting with grade 2 or 3 soiling [5]. 1.4. Statistical analysis Data were entered into an SPSS (Chicago, IL) 13.0 system. Student t test was used to compare the mean age at operation, operative time, postoperative hospital stay and sacral ratio between the groups. Chisquare tests were applied to compare the morbidities of postoperative complications and clinical results between the two groups. p b 0.05 was considered being statistically significant. Table 1 Demographic features of ARM children undergone LAARP versus PSARP.

Age at operation Classification Sacral Ratio Operative time (h) Postop hospital stay (days)

Recto-prostatic Recto-vesical

LAARP(n = 32)

PSARP(n = 34)

6.5 (3–9) months 18 (56.3%) 14 (43.7%) 0.56 ± 0.14 1.62 ± 0.40 5.8 ± 0.65

6.9 (3–12) months 17 (50%) 17 (50%) 0.60 ± 0.16 2.13 ± 0.30 8.4 ± 0.67

The demographic features of the two groups were summarized in Table 1. There was no statistically significant difference in the age at the time of operation, classification of anomaly types and sacral ratio between the two groups. The mean operative time in the LAARP group was significantly shorter than that of the PSARP group (P b 0.01). The postoperative hospital stay in the LAARP group was significantly shorter than that in the PSARP group (P b 0.01) (Table 1). MRI showed a centrally placed rectum within the muscle complex in all patients of the two groups (Fig. 2). Complications were more common in PSARP group, including wound infections and dehiscence (11.8% vs 0%), rectal retraction (5.9% vs 0%), recurrent fistula (11.8% vs 0%) and anal stenosis (5.9% vs 3.1%) (Table 2). No patient experienced urethral diverticulum in either group according to voiding cystourethrogram and MRI (Fig. 3). The appearance of neo-anus after LAARP is adequate (Fig. 4). Rectal prolapses were found in 3 (9.4%) of 32 after LAARP and none of the patients after PSARP. In the LAARP group, 24 patients were followed up more than 3 years, the median follow up period is 7.5 years (range 4–11). Thirtyfour patients were successfully followed up in the PSARP group. The median followed up period is 15.5 years (range 11–20). There were eleven patients (45.8%) with SR below 0.6 (range, 0.31–0.58) in the LAARP group and 13 patients (38.2%) in the PSARP group (range, 0.28–0.57). There was no significant difference of the distribution between the two groups. The rates of voluntary bowel movement, soiling (grades 1, 2 & 3) and grade 1 constipation were similar in both groups. However, grade 2 or 3 constipation was more frequent in PSARP group for patients with recto-prostatic fistula (35.3% vs 0%, P b 0.01) while there was no significant difference for patients with recto-vesical fistula (Tables 3, 4). When the outcomes according to SR were compared, there was no significant difference between LAARP and PSARP groups in both rectoprostatic fistula and Recto-vesical fistula patients (Table 5). Table 2 Postoperative complications in LAARP and PSARP patients.

P 0.84 0.82 0.49 0.43 b0.01 b0.01

Wound infections/dehiscence Rectal retraction Recurrent fistula Urethral diverticulum Anal stenosis Rectal prolapse Overall

LAARP (n = 32)

PSARP (n = 34)

P

0 0 0 0 1 (3.1%) 3 (9.4%) 4 (12.5%)

4 (11.8%) 2 (5.9%) 4 (11.8%) 0 2 (5.9%) 0 12 (35.3%)

0.04 0.15 0.04 – 0.56 0.08 0.04

562

A.-X. Ming et al. / Journal of Pediatric Surgery 49 (2014) 560–563 Table 3 Functional results of patients with recto-prostatic fistula.

Age (years) Sacral Ratio Voluntary bowel movements Free from soiling or grade 1 Soiling grade 2 Soiling grade 3 Constipation grade 1 Constipation grade 2 or 3

Fig. 3. Voiding cystourethrogram.

3. Discussion While posterior sagittal anorectoplasty (PSARP) is still the gold standard for the surgical management of most ARM with rectobladderneck and recto-prostatic fistula [6,7], LAARP is becoming an increasingly common approach in managing patients with rectobladderneck and recto-prostatic fistula ARM. The benefits of LAARP include minimal perineal dissection, excellent visualization of the rectal fistula and surrounding structures, and preservation of the distal rectum with accurate placement of the rectum within the levator ani and external anal sphincter muscle complex [8]. In recent years, controversy remains as to whether LAARP offers functional advantage [9]. Our results showed that operative time and postoperative hospital stay in the LAARP group were significantly shortened. LAARP is obviously less invasive than PSARP for HARM. A shorter post-operative stay and less intraoperative bleeding were reported in other studies [10,11]. Some studies revealed that laparoscopy for ARM is a less invasive procedure when compared with those operations that would have previously required a laparotomy (recto-bladder neck fistula). In

LAARP (n = 13)

PSARP (n = 17)

P

7.5 (4–11) 0.64 ± 0.15 92.3% (12/13) 92.3% (12/13) 7.7% (1/13) 0 7.7% (1/13) 0

15.5 (11–20) 0.62 ± 0.16 76.5% (13/17) 76.5% (13/17) 23.5% (4/17) 0 17.6% (3/17) 35.3% (6/17)

b0.01 0.65 0.36 0.36 0.36 – 0.613 b0.01

cases of recto-prostatic fistulae, the laparoscopic approach is feasible and avoids a lengthy posterior sagittal incision [9]. Recurrent fistula occurred in 4 patients in PSARP group, which may be due to inadequate rectum mobilization, leaving the anterior wall under tension. In the procedure of LAARP, the rectum was mobilized completely and gained significant length provided the rectal wall was not injured. Precise understanding of anatomic relationships can help to avoid injuries to bladder neck, urethra, or an ectopic ureter in the procedure of LAARP. Rectal prolapse was found in 9.4% (3/32) of patients after LAARP and none in patients who underwent PSARP. The incidence of rectal prolapse was higher in LAARP patients. Other studies have shown that rectal prolapse occurs following PSARP, with an incidence of 3%. It is more common in patients with higher malformations and with poor sacral and pelvic musculature [12]. Rectal prolapse have been reported in 8.8%–46% of patients following LAARP [13,14]. In LAARP group, rectal prolapse may be due to the fact that the rectum was inadequately fixed. In order to prevent the morbidity of rectal prolapse, the rectum should be secured to the presacral fascia in the procedure of LAARP, while the dissection of rectum and pelvis should be limited [14]. MRI was used as a tool for the anatomic description of the surgical outcome. It showed a centrally placed rectum within the muscle complex in all cases. Both of the procedures can bring the terminal bowel down to exactly within the sling of the muscle complex. PSARP can significantly improve the visualization of the muscle complex, allowing precise anatomical approximation of the rectum and anus within the reconstructed anorectal anal. LAARP can also provide excellent visualization of the rectal fistula and accurate placement of the pull through segment at the center of the muscle complex. No patient experienced urethral diverticulum in both groups in our voiding cystourethrogram and MRI studies. The fistula was sutured in order to prevent a posterior urethral diverticulum and the mucosa at the end of the fistula was destroyed in the procedure of LAARP. In our experiences, "destruction of mucosa" is very important to preventing recurrent fistula or urethral diverticulum. So the procedure of LAARP can treat the urethral fistula adequately as well as PSARP. Although PSARP procedure exposes the muscles responsible for continence, it divides the constricting mechanism of the muscle complex from coccyx to perineal body. In contrast, LAARP provides an excellent view of the rectal fistula and surrounding structures with Table 4 Functional results of patients with recto-vesical fistula.

Fig. 4. Appearance of neo-anus after LAARP.

Age (years) Sacral Ratio Voluntary bowel movements Free from soiling or grade 1 Soiling grade 2 Soiling grade 3 Constipation grade 1 Constipation grade 2 or 3

LAARP (n = 11)

PSARP(n = 17)

P

7.5 (4–11) 0.52 ± 0.13 72.7% (8/11) 63.6% (7/11) 27.3% (3/11) 9.1% (1/11) 9.1% (1/11) 0

15.5 (11–20) 0.54 ± 0.15 52.9% (9/17) 47.1% (8/17) 35.3% (6/17) 17.6% (3/17) 5.9% (1/17) 11.8% (2/17)

b0.01 0.67 0.435 0.46 1 1 1 0.505

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Table 5 Functional results according to SR.

SR ≥ 0.6

SR b 0.6

Good outcome Poor outcome Good outcome Poor outcome

Recto-prostatic fistula (n =30)

Recto-vesical fistula (n = 28)

LAARP (n = 13)

PSARP P (n = 17)

LAARP PSARP P (n = 11) (n = 17)

10/10 (100%) 0

12/13 (92.3%) 1/13 (7.7%) 1/4 (25%) 3/4 (75%)

2/3 (66.7%) 1/3 (33.3%)

1

2/3 (66.7%) 1 1/3 (33.3%) 0.486 5/8 (62.5%) 0.486 3/8 (37.5%)

6/8 (75%) 2/8 (25%) 2/9 (22.2%) 7/9 (77.8%)

bladder

1 1

The end of rectum 0.153 0.153

less invasion (Fig. 5). The increased association of wound infections after PSARP might be due to the posterior sagittal incision. In the LAARP group, two patients experienced slight constipation and could be managed by diet control, without medication. No patients suffered severe constipation. The outcome was much better than those of the patients in PSARP group, in which grade 2 or 3 constipation was found in 35.3% of patients with recto-prostatic fistula. One possible explanation is that the levator muscle complex was not incised and there was no subsequent post-operative scarring following the procedure of LAARP. Comparing the voluntary bowel movements and soilings, there was no statistically significant difference between the two groups. However, the average age of the patients in LAARP group was lower than that in PSARP group. As functional results improve with time, LAARP appears to provide better outcomes. Studies comparing fecal continence with evaluation questionnaire after LAARP and PSARP in short series showed similar outcomes [10,13,15]. In the study by Lin et al. [16], patients repaired with LAARP had more favorable findings with regard to anorectal manometry than patients repaired with PSARP. There were several drawbacks in our study. Firstly, the study is a retrospective study and not a prospective randomized trial. Hence the results may have confounding bias. Secondly, the average age of the patients in LAARP group was lower than that in PSARP group. This may affect the development of anal sphincter function. Finally, we have just received clinical evaluations for functional results in the follow-up, other objective evaluation methods were not used. Clinical evaluation is important to justify the functional outcomes after the procedure of anorectoplasty, but evaluation of bowel function may be biased because the information concerning the functional outcome is mainly derived from parents. We conclude that LAARP is a less invasive procedure compared with PSARP. The long-term outcomes of LAARP is as good as, if not better than, those of PSARP for children with HARM. References [1] Willital GH. Endosurgical intrapuborectal reconstruction of high anorectal anomalies. Pediatr Endosurg Innov Tech 1998;2:5–11.

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