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Managing Hirschsprung's Disease without Routine Use of Post-Operative Anal Dilatation: Our Experience

Anand Upasani, Thomas Tsang

Department of Pediatric Surgery, Norfolk and Norwich University Hospital, United Kingdom

 

Correspondence:

Anand Upasani

C/O Pediatric Urology Secretaries

Level 7, Southwood Building, Great Ormond Street Hospital

Great Ormond Street, London, UK , WC1N 3JH

Phone : 020 7405 9200

Fax: 020 7813 8260

E-mail : This email address is being protected from spambots. You need JavaScript enabled to view it.

 

Abstract

Background: The current role of anal dilatation following pull-through for Hirschsprung’s disease is ambiguous and subjective. Regular postoperative anal dilatation program is widely practiced. It is unnecessary in all cases and can have potential physical as well as psychological side effects. This is a retrospective review of postoperative course and complications after pull-through procedure with particular reference to the need for routine postoperative anal dilatation.

Material and Methods: We retrospectively, reviewed the cases operated by a single surgeon over a period of 15 years. Postoperatively, all children were continued on low residue feeds. Features suggestive of early enterocolitis or gut dysmotility were used as criteria for instituting anal dilatation. The caliber of the anastomosis was not used as the deciding factor. Twenty case notes were reviewed with regards to postoperative course, use of daily anal dilatations and complications.

Results: Total 20 pull-through procedures were reviewed. Seventeen were males and 3 females. Enterocolitis was observed in 7 (35.0%) of the cases and constipation in 3 (15.0%). Considering the group without daily anal dilatation (n=15), only 2 (13.3%) had significant constipation and 2 (13.3%) developed enterocolitis, whereas all 5 children receiving daily anal dilatation experienced enterocolitis in the early course of management.

Conclusion: It is safe to manage pull-through patients without routine use of post-operative daily anal dilatation. In our cohort there was no anastomotic stricture. Enterocolitis and constipation were comparable to that reported in literature.

Keywords: Hirschsprung’s Disease, pull-through, anal dilatation

 

Introduction

After a pull-through procedure for Hirschsprung’s disease, children are conventionally placed on a program of regular anal dilatation. It is considered that daily dilatation helps in preventing stricture formation and thus reduces constipation and postoperative enterocolitis [1, 2]. However, there have been reports raising concerns of physical as well as psychological side effects from postoperative regular anal dilatation and that it may not be necessary in all cases [1, 3 - 5]. We reviewed our practice of selective postoperative anal dilatation in patients with Hirschsprung’s disease in particular, and report the postoperative course and complications after endo-anal pull-through in our cohort.

Material and Methods

This is a retrospective review of 20 consecutive patients with the endo-anal pull-through performed for Hirschsprung’s disease by a single surgeon over a period of 15 years. Preoperatively, all cases received rectal washouts and dietary modulation with low residue feeds. In the early infant age group, the preferred feeds would be breast milk or a hydrolysed formula (Pregestimil). Intra-operatively, endo-anal dissection was started 5 mm above the dentate line. Submucosal dissection was done up to the peritoneal reflection. Further mobilization of the colon was done 5 cm beyond the last ganglionated biopsy. The sero-muscular sleeve was divided posteriorly and a wide, tension free, hand sewn colo-anal anastomosis was done. Post-operatively, all children were continued on low residue feeds for 6 weeks. Anal calibration was done 2 weeks post-operative. Size or caliber of the anastomosis was not used as criteria for starting anal dilatation. Daily anal dilatation was used postoperatively only in cases with: (1) Unsatisfactory stooling and suspicion of early enterocolitis and (2) Learning difficulties and syndromic association, foreseeing general problems with gut dysmotility.

Medical notes were reviewed with regards to postoperative course and complications. Daily anal dilatation was initiated in 5 children 2 weeks postoperatively. Two of them had associated Trisomy 21, 1 had learning difficulties but no syndromic association, 1 had stooling difficulties and early onset enterocolitis and 1 was a redo procedure following previous Duhamel pull-through. One primary endo-anal pull-through had perforation in the immediate postoperative period secondary to fulminant enterocolitis requiring an exploratory laparotomy and colostomy. It was diagnosed to have pull-through of the transition zone on repeat biopsy. A redo staged procedure was then performed after which the child recovered well.

Results

Of 20 patients with endo-anal pull-through procedure reviewed, 17 were males and 3 were females. Definitive surgery was done at a median age of 5.5 months with a range of 1 month to 4 years 5 months. Level of aganglionosis was in the rectum (3), sigmoid colon (10), descending colon (3), transverse colon (2) and total colonic (2). 10 children had a single stage procedure, 9 had two stage procedures and one was an endo-anal pull-through following failed Duhamel procedure. The median follow-up was 75 months with a range of 8 months to 15 years (Table 1).

Fifteen children had satisfactory progress in the early postoperative period and daily anal dilatation was deemed not necessary. Overall enterocolitis was observed in 7 (35.0%) of the cases and constipation in 3 (15.0%). One of the patients, with total colonic Hirschsprung’s disease, had mild nocturnal soiling. There were no cuff-abscesses or anastomotic strictures noted. (Table 1) Considering the group without daily anal dilatation (n=15), only 2 (13.3%) had significant constipation and 2 (13.3%) developed enterocolitis, whereas all 5 children receiving daily anal dilatation experienced enterocolitis in the early course of management.

Table 1 - Nature of operation and complications

 

 

Endo-anal pull-through (Total cases =20)

          One stage

         Two stage

Redo

Lap-assisted (9)

Open

 

(1)

Lap-assisted  (2)

Open

 

(7)

Lap-assisted

Open

 

(1)

Anal dilatation required (5)

2

-

1

1

-

1

Constipation ( 3 )

2

-

-

1

-

-

Enterocloitis (  7  )

3

-

1

2

-

1

True incontinence (1 )

-

-

-

1

-

-

Cuff abscess (0)

-

-

-

-

-

-

Anastomotic stricture (0)

-

-

-

-

-

-

Discussion

The reported incidence of Hirschsprung’s associated enterocolitis (HAEC) varies from 4.6-54% [6]. Teitelbaum et al. have reported an incidence of 42% of postoperative enterocolitis with single stage procedure [7]. Down’s syndrome has been identified as a significant risk factor for HAEC [8]. In our study HAEC was seen in 7 (35.0%) children. Our rate of HAEC is comparable to the above studies and 2 of the 7 (28.5%) had Down’s syndrome. Constipation was seen in 3 (15.0%) of our 20 cases. This is comparable to other studies which report an incidence of 28.0% [7]. Anastomotic complications, particularly stricture or leak are reported with an incidence of 4.0 -22.0% [1]. None of our patients had anastomotic complications.

Postoperative daily anal dilatation is widely used following pull-through procedures for anorectal malformations and Hirschsprung’s disease. The current role of regular anal dilatation following endo-anal pull-through is ambiguous and subjective. Individual opinions reflect local practices rather than being evidence based. It has been considered that routine anal dilatation stimulates rectal growth and stretches gently the muscles preventing rupture. It has also been reported that daily dilatation helps in preventing stricture formation and thus reduces constipation and postoperative enterocolitis [1, 2].

There are also adverse effects perceived in carrying early post-operative anal dilatation. Disruption of the anastomosis has been reported following anal dilatation [1]. Theoretically, it can accentuate the post-operative edema of the ano-rectal wall and may introduce infection leading to enterocolitis. Daily anal dilatation is psychologically stressful to both the parents and the child [1]. Studies have shown that invasive anal procedures can negatively affect the children and their careers. Children undergoing regular anal dilatation are more prone to mental health and psycho-social problems as compared to general population. Diseth [3-5] has demonstrated that anal dilatation in first 4 years of life of a child is a significant predictor of dissociative experience in adolescence and adulthood.

Temple et al. [1] have attempted to reduce the dilatations from daily to weekly basis and suggested that it should be done by the medical professionals rather than the parents to reduce parental stress and minimize the chances of anastomotic dehiscence. Obermayr et al. [9] have used postoperative narrowing of the anastomosis as the criteria for initiating anal dilatation. We believe in the early stage after the pull-through, low residue feeds will allow easier regular bowel emptying. The loose to soft consistency of stools put less stress on the inflamed anastomotic tissues but gives the lumen a gentle endogenous dilatation. This seems very effective in the majority of patients who did not have routine daily anal dilatation in our series. Early planned routine anal dilatation did not seem to stop or reduce the enterocolitis in the selected 5 patients. It is understood that HAEC is a multi-factorial condition with the impaired colonic mucosal defense playing a significant role which may or may not be helped by the routine anal dilatation. The latter has been reported to prevent or reduce anastomotic strictures, but in the present study none of the children developed anastomotic stricture which includes 15 (75.0%) cases, in whom daily anal dilatation was not used [7, 9, 10]. In our limited experience, without the use of routine anal dilatations there was no difference in the outcome as compared to the literature.

Conclusion

It is safe to manage pull-through patients without routine use of post-operative daily anal dilatation. In our cohort there was no anastomotic stricture. Enterocolitis and constipation were comparable to that mentioned in literature.

 

 

References:

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5. Diseth TH, Egeland T, Emblem R. Effects of anal invasive treatment and incontinence on mental health and psychosocial functioning of adolescents with Hirschsprung's disease and low anorectal anomalies. J Pediatr Surg 1998; 33:468-75.

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9. Obermayr F, Szavay P, Beschorner R, et al. Outcome of transanal endorectal pull-through in patients with Hirschsprung's disease. Eur J Pediatr Surg 2009; 19:220-3.

10. Rouzrokh M, Khaleghnejad AT, Mohejerzadeh L, et al. What is the most common complication after one-stage transanal pull-through in infants with Hirschsprung's disease? Pediatr Surg Int 2010; 26:967-70.