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 Table of Contents  
ORIGINAL ARTICLE
Year : 2019  |  Volume : 5  |  Issue : 2  |  Page : 131-134

Primary management of anorectal malformation: Initial result at a tertiary care centre


1 Additional professor, Dept. of Paediatric Surgery, IGIMS, Patna, Bihar, India
2 Professor & Head, Dept. of Paediatric Surgery, IGIMS, Patna, Bihar, India
3 Associate professor, Dept. of Paediatric Surgery, IGIMS, Patna, Bihar, India
4 Assistant Professor, Dept. of Paediatric Surgery, IGIMS, Patna, Bihar, India

Date of Submission24-Jun-2019
Date of Acceptance15-Jul-2019
Date of Web Publication20-Nov-2020

Correspondence Address:
Vinit Kumar Thakur
Additional professor, Dept. of Paediatric Surgery, IGIMS, Patna
India
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Source of Support: None, Conflict of Interest: None


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  Abstract 


Background : Anorectal malformations [ARM] are common neonatal surgical emergencies. They can be treated either by staged procedure or by single-stage procedure. In the present study, we have performed single-stage surgery of ARM in both male and female.
Materials and Method: Retrospective review of cases of ARM operated over 3 years in the Department of Paediatric surgery at a tertiary care centre from January 2015 to December 2018 was done. In this study, we compared the results of single stage repair of Anorectal malformation (category1) with that of staged procedure performed earlier (category 2).In male babies 43 patients were operated by primary PSARP (posterior sagittal anorectoplasty) and remaining 11 by primary APP (abdomino-perineal pull through) procedure. Out of 65 female patients, PSARP was performed in 39 (60%) cases of recto vestibular fistula, ASARP in 23 (29.2%), Primary Abdomino-perineal pull-through in 4 (6.1%) cases of common cloaca. The result of this procedure was compared with that of staged procedure. (Category 2)
Result : Mean post- operative hospital stay in category 1 was 11 days and 32 days in category 2. Mean duration of surgery in category 1 was 1hour and 15 minutes and 3 and half hours in category 2 that included initial colostomy, PASRP or APP and colostomy closure. In category 1, 41 (75.9%) cases had stool frequency 3- 5 per day while in category 2, 20 (51%) patients had the same frequency at the age of three years which was found to be statistically significant (P value? 0.001). Similar findings were observed in female patients.
Conclusion : Single-stage surgical repair of Anorectal malformation in both male and female patient are reliable and can be safely performed with advancement of surgical expertise and better anaesthetic support with few exceptions. Early results are encouraging. However, long term study is required for its justification.

Keywords: Anorectal malformation; Male; Female; Primary


How to cite this article:
Hasan Z, Kumar V, Yadav R, Rahul SK, Chaubey D, Prasad R, Thakur VK. Primary management of anorectal malformation: Initial result at a tertiary care centre. J Indira Gandhi Inst Med Sci 2019;5:131-4

How to cite this URL:
Hasan Z, Kumar V, Yadav R, Rahul SK, Chaubey D, Prasad R, Thakur VK. Primary management of anorectal malformation: Initial result at a tertiary care centre. J Indira Gandhi Inst Med Sci [serial online] 2019 [cited 2021 Jan 18];5:131-4. Available from: http://www.jigims.co.in/text.asp?2019/5/2/131/301096




  Introduction : Top


Anorectal malformations (ARMs) are birth defects with absent or malformed anus. Its incidence occurs in 1 in 2500 to 5000 births but may be frequent in certain developing countries[1]. Traditionally in Anorectal malformation, neonatal diverting colostomy followed by PSARP/APP and subsequent colostomy closure is done. However, primary surgery without using colostomy is an emerging trend as colostomy is often associated with morbidity and mortality and generally not favoured by parents in the modern society aesthetically. Keeping this in mind, we have performed primary surgery for Anorectal malformation.


  Materials and Method : Top


Retrospective review of cases of ARM managed from January, 2015 to December, 2018 was undertaken. In (Category 1), total of 54 male and 65 female patients of Anorectal malformation were operated in single stage from January, 2015 to December, 2018. The results of this procedure were compared with that of staged procedure (Category 2) in which a total of 39 male and 29 female were operated before 2015. Routine blood count, biochemistry, renal function test, Ultrasonography of abdomen and echocardiography were performed in all patients along with Prone cross table lateral X-ray with focus on greater trochanter for detecting the level of gas shadow. If the gas shadow was above PC line we did Abdomino-perineal pull through procedure (APP) procedure and if found below PC line we did posterior sagittal anorectoplasty (PSARP) procedure. We excluded low ARM and female babies presenting with ARM. Out of 54 patients of ARM, 43patients were operated by PSARP (posterior sagittal anorectoplasty) for intermediate ARM and 11 by APP (Abdomino-perineal pull through) for high ARM procedure. In two patients, Abdomino-PSARP procedure was done because pouch could not be located by PSARP approach. In these cases, we divided the terminal pouch as far down using bipolar cautery and left it open without ligating. In category 1, female patients PSARP was performed in 39 (60%) cases of recto vestibular fistula, ASARP in 23 (29.2%), Primary Abdomino-perineal pull-through in 4 (6.1%) cases of common cloaca. In 2 (3%) cases, we did PSARP in H- type fistula and in 1(1.5%) case we performed PSARP in intermediate ARM without fistula in a baby with Down’s syndrome. We excluded following patients presenting with Anorectal malformation.

  1. Low ARM
  2. Presenting with gut perforation
  3. Associated with complex anomalies



  Procedure: Top


All the surgeries were done under general anaesthesia with endotracheal intubation. Primary PSARP procedure was done using midline posterior sagittal incision after taking stay suture over the proposed anus. Muscle and sphincter was cut in the midline using needle tip cautery. We first deflate the air from terminal pouch using syringe simultaneously we avoid using Babcock forcepas it is crushing to nascent bowel rather we use stay suture and vascular forceps to hold the bowel. We also use wet gauge piece for mobilising the rectal pouch. Terminal pouch was opened and meconium sucked out; subsequent submucosal dissection was done proximal to fistula site and pouch mobilised. Anorectoplasty was done after division and closure of fistula. In APP procedure; abdomen was opened by left lower paramedian incision. Bowel was deflated and traction to terminal pouch was given using infant feeding tube, dissection of rectum begun at the site of peritoneal reflection ligation of fistula done, and subsequent pull-through of colon followed by anoplasty. In female patients, bowel is prepared using rectal wash with saline glycerine. During surgery, we utilise double Foleys catheter one in urethra and another inside rectum for better localisation of rectum. We do not cut fourchette. In the Post- operative period, patients were nursed in surgical intensive care unit in prone position to prevent faecal contamination. Post- operative dressing was done using diluted povidone iodine and mupirocin ointment. Patients were advised Hegar dilatation after two weeks of surgery.


  Result : Top


In category 1, mean age of presentation was 3.05 days. Among the 54 male cases of ARM, we had 43(79.6%) cases of Intermediate ARM and 11(20.3%) cases of high ARM. [Table 1]. Among 11 patients of high ARM, 4 patients were of pouch colon. Mean duration of surgery in category1 was 1hour and 15 minutes and 3 and half hours in category 2 that includes initial colostomy, PASRP or APP and colostomy closure. In category 1, post- operative feeding was started from 3rd (mean) post-operative day in male and from 2nd day in female. Urinary catheter was removed by 7-8 days post operatively.Mean post- operative hospital stay in category 1 was 11days and 32 days in category 2. The difference was found to be significant statistically. In post-operative period, in both categories patients were kept on enema to keep the rectum empty which was required less frequently in category 1infollow up visit. Mean weight gain for the corresponding age was better in category 1 than category 2. In the patients of high ARM, there was frequent defecation and associated perineal excoriation which lessened in the subsequent visits on conservative management. In eight patients (14.8%) in category 1 there was perineal excoriation and five patients suffered from minor wound infection. [Table 3] Similar results were found in female patients. [Table 4] They were managed using dressing and zinc oxide based ointment. In category 1, 41 (75.9%)cases had stool frequency 3-5 per day while in category 2, 20 (51%) patients had the same frequency at the age of three years which was found statistically significant (p-value? 0.001). Similarly in female patients, 51(78.4%) had stool frequency 3-5 per day while in category 2, 14 (48%) at the end of 3 years of follow up. Various complications associated with both procedures in both male and female are shown in the [Table 3] & [Table 4] in which perineal excoriation and wound infection were encountered more in category 1 than in category 2 in both male and female patients. Perineal excoriation was encountered mainly in high ARM patients. In two patients in category 1 there was wound disruption requiring secondary suturing. Mucosal prolapse was detected in 1 case in the category 1 and 2 cases in the category 2 There was one death in category 1 in the post-operative period due to sepsis whereas in category 2 there were two death due to colostomy related complication.
Table 1: Types of Male ARM with their percentile and procedure

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Table 2: Types of female ARM with their Percentile and procedure

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Table 3: Post-operative Complications in Male patients of Category 1 and Category 2

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Table 4: Post- operative complication in female patients in category 1 and 2

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  Discussion : Top


ARM have been a source of concern for many centuries Incidenceof ARM occurs in 1 in 2500- 5000 births but may be frequent in certain developing countrieswith higher preponderance in boys.[1] The etiology of such malformations remains unclear and is likely multifactorial. The treatment of ARM mainly consists of anoplasty,PSARP, abdomino-perinealpull-through for the selected group of high ARM. Recently, Georgeson et,al.[2] employed a laparoscope-assisted one-stage AP pull-through procedure that provided excellent exposure deep in the pelvis. Selecting the one-step or three-step surgery to treat this condition has been a subject of debate for many years. Reasons for choosing the 3-stage repair are the lesser risk of surgery because of faecal diversion by colostomy. It might be an easier surgical technique because of the delay in definite repair and weight gain by the infant. On the other hand, the reasons for choosing one-stage repair are multiple, for example, avoidance of multistage operations and saving time and costs, less morbidity for children and avoidance of all colostomy related complications. In addition dissection is easier and takes less time in the neonatal period due to virgin tissue planes.[3] Moreover, children with colostomy will have problems in peer relationships. There would be school absentees and behavioural problems. This led us to evolve single-stage management of all ARM cases. Definitive procedure in the neonatal period itself is recommended as cerebral cortical fibers develop in the first few year of life and sensation of rectal fullness are essential for these fibers to develop fully so that continence can be achieved to its maximal potential.[4],[5],[6] Several authors in literature have reported single-stage repair of ARM cases by PSARP in neonates to be safe and feasible in selected group of patients.[7],[8],[9] Elhalabyet,al in their study on 38 patients demonstrated that primary repair of high and intermediate ARMs in infants were feasible.[10] Another study in 2005, conducted by Gangopadhyayet al, in 105 patients, they found that an initial one-stage surgery method had better apparent results and fecal control was associated with a reduced mortality rate and reduced cost.[11] The safety of the approach depends on the exclusion of associated anomalies with anorectal malformation.[12] The normal pattern of defecation in children in the 1st month of life is about 6 times/day, and after 2 months, decreases to 1-2 times/day until the child is 2 years old. [13] In female patients of rectovestibular fistula which constituted bulk of our patient (60%), we have utilised experience of Upadhyay et, al 18 in single stage repair witout opening of fourchette. The clinical scoring methods were devised by Kelly, Templeton et al, Kiesewetter and Chang, and Stephens and Smith according to the degree of continence and the quality of life after management.[14] Many studies have been carried out to assess the continence results after PSARP and compare it with other methods in order to prove the superiority of one method over another. Rintala and Lindahl reported PSARP to be superior to sacroperineal and sacro-abdominoperineal pull-through in terms of long- term bowel function and faecal continence.[15] On the contrary, Gil-Vernet et al. found better results with abdominoperineal pull-through plus PSARP in comparison to PSARP in patients with high defects.[16] In our study category 1 has better continence result as compared to category 2 as the definitive procedure was done very early, so that anorectal continuity could be achieved early. The early post-operative complications in the presentstudy are acceptable and its functional results are better with that of conventional multi-stage procedure.We find full justificationwith Alberto Pena’s comment that “we should all move in the direction of repairing anorectal anomalies earlier and in a single operation.”[17]


  Conclusion : Top


This is a comprehensive study of primary management of Anorectal malformation at a tertiary care centre involving both male and female patient. One-stage surgical repair of Anorectal malformation both male and female patient are reliable and can be safely performed with advancement of surgical expertise and better anaesthetic support with few exceptions. Early results are encouraging and hope is virtual elimination of colostomy in almost all cases of anorectal malformation however, long term study is required for its justification.

Legends to the Table

Table 1 Types of Male ARM with their percentile and procedure

Table 2 Types of female ARM with their Percentile and procedure

Table 3 Post-operative Complications in Male patients of Category 1 and Category 2

Table 4 Post- operative complication in female patients in category 1 and 2



 
  References Top

1.
Holschneider AM, Hutson JM 2006;Anorectal Malformations in Children: Embryology, Diagnosis, Surgical Treatment.In:Moore SW(ed) Genetics,Pathogenesis and epidemiology of anorectal malformation and caudal regression syndrome,1stedn springer pp3.  Back to cited text no. 1
    
2.
Georgeson KE, Inge TH, Albanese CT. Laparoscopically assisted anorectal pull-through for high imperforate anus 2000; new technique. J PediatrSurg 35:927-30.  Back to cited text no. 2
    
3.
Gangopadhyay AN, Gopal SC, Sharma S, Gupta DK, Sharma SP, Mohan TV.2005; Management of anorectal malformations in Varanasi, India: a long-term review of single and three stage procedures. PedaitrSurg Inter 22:169-72.  Back to cited text no. 3
    
4.
Albanese C, Jennings RW, Lopoo JB et al 1999; One stage correction of high imperforate anus in the male neonate. J PediatrSurg 34:8346.  Back to cited text no. 4
    
5.
Dobbing J, Sands J 1973; Quantitative development of growth and development of the human brain. Arch Dis Child 48:757.  Back to cited text no. 5
    
6.
Moore TC 1990; Advantages of performing the sagittal anoplasty operation imperforate anus at birth. J PediatrSurg 25:276.  Back to cited text no. 6
    
7.
Elsaied A, Aly K, Thabet W, Magdy A 2013; Two-stage repair of low anorectal malformations in girls: Is it truly a setback? Ann PediatrSurg 9:69-73.  Back to cited text no. 7
    
8.
Osifo OD,Osagie TO, Udefiagbon EO 2014; Outcome of primary posterior sagittal anorectoplasty of high anorectal malformation in well selected neonates.Niger J ClinPract; 17:1-5.  Back to cited text no. 8
    
9.
Liu G,Yuan J, Geng J, Wang C, Li T. 2004;The treatment of high and intermediate anorectal malformations : one stage or three. J of pediatrsurg; 39:1466-71.  Back to cited text no. 9
    
10.
Elhalaby EA 2006; Primary repair of high and intermediate anorectal malformations in the neonates. Ann PediatrSurg 2:117-22.  Back to cited text no. 10
    
11.
Gangopadhyay AN, Shilpa S, Mohan TV, Gopal SC 2005; Single-stage management of all pouch colon (anorectal malformation) in newborns. J Pediatr Surg 40:1151-5.  Back to cited text no. 11
    
12.
Gangopadhyay AN, Pandey V 2017; Simultaneous single-staged repair of anorectal malformation with tracheoesophageal fistula: Lessons learned. J Indian AssocPediatr Surg.;22:96-100.  Back to cited text no. 12
    
13.
Adeniran JO 2002; one-stage correction of imperforate anus and rectovestibular fistula in girls: Preliminary results. J PediatrSurg 37:16.  Back to cited text no. 13
    
14.
Ong NT, Beasley SW 1990; Comparison of clinical methods for the assessment of continence after repair of high anorectal anomalies. PediatrSurg Int. 1; 5:233-7.  Back to cited text no. 14
    
15.
Rintala RJ, Lindahl HG 1999; Posterior sagittal anorectoplasty is superior to sacroperineal-sacroabdominoperinealpullthrough: a long-term follow-up study in boys with high anorectal anomalies. J PediatrSurg 34:334-337.  Back to cited text no. 15
    
16.
Gil-Vernet JM, Asensio M,Marhuenda C, Broto J, Wayar A 2001; Nineteen years’experience with posterior sagittal anorectoplasty as a treatment of anorectal malformation. Cir Pediatr 14:108-11.  Back to cited text no. 16
    
17.
Albanese C, Jennings RW, Lopoo JB et al. One stage correction of high imperforate anus in the male neonate. J PediatrSurg 1999; 34: 834-836.  Back to cited text no. 17
    
18.
Upadhyay VD, Gangopadhyay AN, Pandey A, Kumar V, Sharma SP. 2008; Single stage repair of rectovestibular fistula without opening of fourchette. J of PediatSurg 43;775-9.  Back to cited text no. 18
    



 
 
    Tables

  [Table 1], [Table 2], [Table 3], [Table 4]



 

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