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ORIGINAL ARTICLE
Year : 2018  |  Volume : 23  |  Issue : 1  |  Page : 4-9
 

Anterior sagittal anorectoplasty with external sphincter preservation for the treatment of recto-vestibular fistula: A new approach


Department of Surgery, Pediatric Surgery Unit, Tanta University Hospital, Tanta, Egypt

Date of Web Publication27-Dec-2017

Correspondence Address:
Mohamed Ibrahim Elsawaf
Department of Surgery, Pediatric Surgery Unit, Tanta University Hospital, Tanta
Egypt
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jiaps.JIAPS_2_17

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   Abstract 

Context: To our knowledge, there is no description of anterior sagittal anorectoplasty (ASARP) with external anal sphincter preservation and passing neorectum in the middle of muscle complex under direct vision for the treatment of recto-vestibular fistula (VF).
Aim: This study evaluates a new modification combining ASARP with under vision sphincter preservation.
Subjects and Methods: This prospective study was conducted on thirty female infants with VF. Procedure starts with a vertical midline incision extending from ectopic opening to posterior limit of external sphincter. Sharp dissection of the fistula is carried out. Artery forceps is passed at the center of muscle complex under vision, then neorectum is placed in the middle of the muscle complex. We introduced a new scoring system based on parental interview assessing functional outcome. Each patient was given a score between 0 and 20; good: 14–20, fair: 7–13, and poor: 0–6.
Results: ASARP was performed at a mean age of 2.6 months, a mean weight of 5.2 kg, a mean operative time of 102 min, and a mean hospital stay of 3.6 days. Wound infection occurred in four cases, seven cases had perianal excoriations, six cases had anal stenosis, and only one case complained of anal displacement. Patients followed for a mean of 18.8 months. Majority of our patients (50%) had good score (mean = 16.8), normal frequency, no or mild soiling, normal anal position with no or mild stenosis. Fourteen patients had fair score (mean = 10.5). Only one had poor outcome with severe soiling and perineal excoriation.
Conclusion: Our modification offers optimal correction, with minimal sphincteric damage, without additional complexity or difficulties. Scoring system is simple, practical, and truly reflects early functional and parent satisfaction after surgery.


Keywords: Anorectal malformation, anterior sagittal anorectoplasty, continence scoring, recto-vestibular fistula, sphincter preservation


How to cite this article:
Elsawaf MI, Hashish MS. Anterior sagittal anorectoplasty with external sphincter preservation for the treatment of recto-vestibular fistula: A new approach. J Indian Assoc Pediatr Surg 2018;23:4-9

How to cite this URL:
Elsawaf MI, Hashish MS. Anterior sagittal anorectoplasty with external sphincter preservation for the treatment of recto-vestibular fistula: A new approach. J Indian Assoc Pediatr Surg [serial online] 2018 [cited 2018 Jul 19];23:4-9. Available from: http://www.jiaps.com/text.asp?2018/23/1/4/221591





   Introduction Top


Recto-vestibular anus is the most common anorectal malformation (ARM) in female patients, in which rectum opens immediately behind the hymen in the vestibule.[1] Several surgical techniques are described for its correction including cutback,[2] anal transposition,[3] posterior sagittal anorectoplasty (PSARP),[4] and anterior sagittal anorectoplasty (ASARP), in which anterior rim of the external anal sphincter is cut and puborectalis sling is left untouched.[1],[5],[6],[7] Neutral sagittal anorectoplasty (NSARP)[8] and transfistula anorectoplasty (TFARP)[9] are also techniques described in VF treatment. Wang et al. described modified ASARP using endoscopic magnification to avoid cutting external sphincter.[10]

The rationale of this study is to evaluate a new technical innovation which combines the advantages of ASARP with sphincter preservation and properly positioning rectum at the center of anal sphincter under direct vision. To our knowledge, there is no literature description of such technique.


   Subjects and Methods Top


This is a prospective study on thirty female infants with vestibular fistula (VF) according to the Anatomical and Krickenbeck Classification,[11] presented to us from August 2013 to August 2016. Regular dilatations of fistula using dilator size #7 were carried out to decompress the rectum. Age at operation was between 2 and 4 months. Preoperative bowel preparation is carried out with only clear fluids orally for 24 hours and rectal washouts with saline was performed at 6 hourly intervals until the effluent was clear. All parents signed informed consent.

The patient was put in a lithotomy position with both lower limbs supported on a metal bridge. Vestibule was spread by fixing labia majora on both sides to the thigh using 3/0 silk sutures. Determining the site of a proper anus using Pena stimulator, then marking anterior and posterior limits of external anal sphincters was done using 5/0 silk sutures. Circumferential 6/0 silk sutures were applied to mucocutaneous junction around fistulous opening for traction. We then perform vertical skin incision strictly in midline extending from the ectopic opening to posterior limit of sphincter previously identified; hence, anterior part of external sphincter comes into sight. Sharp dissection of the fistula was carried out with meticulous care not to damage rectal musculature. The common wall between vagina and rectum was meticulously dissected apart with complete separation of rectal tube from vagina to avoid retraction. Bleeding points are controlled by electrocoagulation. We then pass artery forcep in the center of muscle complex deep to its anterior rim without cutting it. We grasp the traction sutures and pass neorectum in the center of the muscle complex. Rectum is fixed afterward to levator ani muscle using 5/0 vicryl to prevent its retraction or prolapse, followed by reconstruction of the perineal body in two layers using 4/0 vicryl, and perineal skin is closed with 5/0 vicryl [Figure 1]. Finally, we cover the wound with antibiotic ointment and apply betadine-soaked dressing.
Figure 1: Modified anterior sagittal anorectoplasty operative step photographs

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Intravenous antibiotics (4th-generation cephalosporin and metronidazole) continued till 3rd postoperative day, followed by oral cephalosporin for 4 more days. Foley catheter was kept in situ for 3 days for better perineal care. We started oral feeding 6–9 h postoperatively. The patients were discharged within 3–4 days of surgery unless otherwise indicated. All patients were scheduled for anal dilatations from 14th postoperative day and followed up for at least 3 months.

We also introduce a new scoring system based on a personal interview with parents or guardians to assess functional outcome. This scoring system is based on a questionnaire which consists of six items. Each patient was given a score ranging from 0 to 20; 14–20 is good, 7–13 is fair, and 0–6 is poor.

Children were evaluated for as follows:

  1. Anal position (anal position index [API] calculated by ratio of anus-posterior fourchette to coccyx-posterior fourchette distance). API <0.34 was defined as anterior displacement [12]
  2. Anal size
  3. Facial expression during defecation
  4. Stooling frequency
  5. Soiling
  6. Perianal excoriation.



   Results Top


Our study included thirty female infants with VF managed by ASARP with external sphincter preservation. Age at operation ranged from 2 to 4 months, with a mean 2.6 ± 0.6 months. While weight at operation ranged from 4 to 7 kg, with a mean 5.2 ± 0.9 kg. Operative time ranged from 90 to 120 min, with a mean 102 ± 11 min. Vaginal injury occurred in 3 patients (10%) and rectal injury in 1 patient (3.33%). Neither major bleeding nor accidental cutting of anterior part of external sphincter occurred. We preserved hymen in all cases. Hospital stay ranged from 3 to 14 days, with a mean 3.6 ± 2 days; only one case stayed for 2 weeks because of wound disruption.

Superficial wound infection occurred in 4 cases (13.3%), which treated by frequent dressing. Only one had wound with disruption which healed by secondary intention. Nine cases (30%) suffered perianal excoriations; all were treated with zinc oxide and topical steroids. Six cases (20%) had anal stenosis, and one case was severely stenotic and required dilatation under general anesthesia, while the other five cases managed with regular daily dilatations.

Patients are followed for 3–34 months, with a median age of 17.5 months and mean age of 18.8 ± 10.4 months. Short-term functional outcomes were evaluated 3-month after operation using our new scoring system. As shown in [Table 1], nine patients (30%) had normal stool frequency referring to good continence, while soiling reported in 21 patients (15 with constipation and six without). Normal anal position in all patients except one who had mild anal displacement according to API. Constipation occurred in 15 patients (50%); diet modification solved the problem in three patients, and regular laxatives had good results in 12 patients.
Table 1: Incidence and distribution of our new score items among our patients

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Majority (15 patients) were included in Group A (good) with score ranging 14–19 and mean 16.8 ± 1.9; they showed normal frequency of defecation, no or mild soiling, normal anal position with no or mild anal stenosis. Fourteen patients were included in Group B (fair) with score ranging between 9 and 13, with mean 10.5 ± 1.73; these patients have constipation, mild to severe soiling, anal excoriation, and mild to severe stenosis. Only one patient was included in Group C (poor) which showed severe soiling, displaced anus, and severe anal excoriation [Table 2].
Table 2: The results of functional outcome score

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   Discussion Top


Several surgical techniques were described for correction of VF. However, wide range of patients still suffers different functional sequelae as constipation, soiling, and even incontinence. Because of the physiological importance of anal muscle complex in continence, we introduce the idea of sphincter saving as a modification of ASARP in the treatment of VF. In anal transposition, rectum is placed in the anal muscle complex blindly.[3] PSARP entails cutting levator ani and external anal complex.[4] Original ASARP entails cutting of anterior rim of external anal sphincter.[5] Endoscopic guidance to preserve external anal sphincter adds complexity to ASARP,[10] while NSARP [8] entails opening the levator ani muscle, tackling fistula from inside the rectum, and nonreconstruction of the perineal body. TFARP [9] is similar to anal transposition without skin incision; it also has the disadvantages of poor reconstruction of the perineal body and blind tunneling of the anal sphincter. To our knowledge, there is no description to combine ASARP with external anal sphincter preservation and passing neorectum in the middle of muscle complex under direct vision.

We believe that early correction of ARM contributes to good continence outcome. Hence, we elected to operate between 2 and 4 months, mean 2.6 months. Furthermore, Wakhlu et al. operated at 3–6 months,[1] while Kuijper and Aronson operated at 0–73 months.[13] Operative complications were minor and comparable to others; vaginal wall injury in 10%, rectal injury only in one case, no accidental cutting of anterior part of external sphincter occurred, and we preserved hymen in all cases. Wakhlu et al. reported vaginal injury as most frequent problem, while rectal injury was uncommon.[1]

Preservation of intact anal sphincter gives extra protection to the wound as infection is a threat by stool contamination, and in the most worst scenarios when wound disruption occurs, there is no impact on continence. This allowed us to avoid using protective colostomy and enabled us to do the procedure as one stage. Furthermore, we were able to start oral feeding 6–9 h postoperatively as reported by Waheeb [7] and Wakhlu et al.[1] On the contrary, oral feeding started on the 4th postoperative day nearly for almost all studies.[7],[13],[14] Wound infection,[1],[13],[14],[15] disruption, and dehiscence requiring redo surgery were reported in many studies with one-stage VF repair.[1],[13],[14] In our study, wound infection occurred in four out of thirty cases. They were treated by frequent dressing. Only one of them had wound disruption and healed with secondary intention.

Rectal prolapse, retraction, and anterior migration of anus were reported in several studies.[6],[14] In our study, these complications did not occur due to adequate mobilization of the rectum from vagina, preservation of sphincter, and fixation of neorectum to muscle complex. The API for all cases was above 0.34 except for one with wound disruption which had mild anal displacement due to large anal size.

Constipation is expected after RV repair, because we preserved the complex muscle and passed the neorectum in a narrow tunnel in its center and we elected not to dilate it intraoperatively to avoid its tear. Half of our patients have more or less normal frequency with good continence, while 15 infants suffered constipation improved with medical treatment or diet modification. Kumar et al. reported constipation in 25.68% of patients, managed by rectal wash, laxatives, and diet modification, but two patients had severe recurrent constipation needed frequent manual disimpaction.[14] Kuijper and Aronson reported constipation in 34% of patients who needed regular laxative and/or enema.[13] We started anal dilatation routinely at the 14th postoperative day to avoid anal stenosis. Kumar et al. reported 6.76% of anal stenosis due to irregular or absence of anal dilatation.[14] Wakhlu et al. reported anal stenosis in five cases.[6] We faced postoperative stenosis in six cases; all were managed at home with regular daily dilatation except for one case with severe anal stenosis that required dilatation under general anesthesia followed by daily regular dilatation at home.

Postoperative soiling was reported in many studies. Javid et al. reported soiling in 47%,[16] 50.8% in Liu et al.,[17] and 66% in Hamid et al.[18] However, other studies did not have soiling as a common postoperative complication. In our study, the majority of soiling (15 cases) occurred with constipation due to retention with overflow and disappeared with diet modification, while the remaining six cases (20%) were due to improper development of anal sphincter or its innervations and improved gradually with time. Perianal excoriation was noted in nine out of thirty (30%) cases due to the presence of soiling. Fortunately, all responded to treatment using zinc oxide and topical steroids within 1–3 months. On the contrary, Kumar et al. reported one case out of 94 had excoriation.[14]

Our modification in the treatment of VF combines the merits of original ASARP from adequate exposure, proper mobilization of rectum up to the sacral promontory, and adequate reconstruction of the perineal body for better cosmoses and function, with preservation of the integrity of external anal sphincter leading to better continence outcome.

Kamal compared functional outcome of different techniques used in repair of VF. In his study, constipation was common in anal transposition and less in ASARP and PSARP, where prolapse, dislocated anus, and ugly scaring were frequently detected in both ASARP and PSARP.[19] Shehata used Templeton and Holschneider scores to assess early and late functional outcomes in primary VF repair and concluded that ASARP is optimal, perineal cosmetic results were superior to PSARP, excellent continence was achieved, and functional results gave higher scores in ASARP as compared to PSARP.[20] Waheeb used clinical continence score proposed by Ditesheim and Templeton to assess continence in children over 3 years and concluded that ASARP promised many advantages in VF.[7]

All present continence scores can be used only after 3 years of age.[21] We introduce a new functional scoring system to provisionally access postoperative continence outcome before the age of 3 years. It is based on a questionnaire consisting of six items assessing bowel habits, anatomical landmarks, physical and clinical outcome to be able to give a clue on the future continence after ARMs correction. This questionnaire uses frequency of defecation and straining in the absence of stenosis as a proof for intact sphincter. Soiling without constipation reflects weak or absent sphincteric control and the presence of excoriation points to soiling in younger age groups. Anatomical landmarks such as anal size and position reflect adequate repair and proper positioning of neorectum in the center of muscle complex. Although we have 14 patients in the fair group, we think this is due to mild anal stenosis in nine patients, soiling in 21 patients, and constipation in 15 patients. In our minds, this occurred due to our reluctance to do intraoperative dilatations to avoid tearing of the preserved anal complex, and also for having the questionnaire after 3 months, by this time, the most of these problems still did not resolve. We do expect the most of them to move to the good group after finishing dilatations and resolution of stenosis and soiling problems. The majority of our patients (29 patients) had good and fair continence score with our new ASARP modification using our newly introduced score. Long-term follow-up and randomized controlled studies are mandatory to confirm our results and to validate our newly introduced scoring system.


   Conclusion Top


We introduce sphincter-saving modification of ASARP procedure in the treatment of VF, combining ASARP with preservation of external anal sphincter and passing neorectum through a central tunnel made in external muscle complex under direct vision. Furthermore, we introduce a new simple scoring system to evaluate functional outcome in babies younger than 3 years of age.

We believe that this new modification offers a lot to VF patients with almost optimal correction of their anomaly, minimal damage to muscle complex, and without adding further complexity or difficulties. The new proposed scoring system is simple, practical, and truly reflects functional and overall parent satisfaction after treatment of ARMs.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Wakhlu A, Pandey A, Prasad A, Kureel SN, Tandon RK, Wakhlu AK. Anterior sagittal anorectoplasty for anorectal malformations and perineal trauma in the female child. J Pediatr Surg 1996;31:1236-40.  Back to cited text no. 1
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de Vries PA. High, intermediate, and low anomalies in the female. Birth Defects Orig Artic Ser 1988;24:73-98.  Back to cited text no. 2
    
3.
Eltayeb A. The anterior sagittal anorectoplasty for the treatment of anovestibular fistula in newborn and children. Ann Pediatr Surg 2008;1:18-21.  Back to cited text no. 3
    
4.
Peña A, Devries PA. Posterior sagittal anorectoplasty: Important technical considerations and new applications. J Pediatr Surg 1982;17:796-811.  Back to cited text no. 4
    
5.
Okada A, Kamata S, Imura K, Fukuzawa M, Kubota A, Yagi M, et al. Anterior sagittal anorectoplasty for recto-vestibular and anovestibular fistula. J Pediatr Surg 1992;27:85-8.  Back to cited text no. 5
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6.
Wakhlu A, Kureel SN, Tandon RK, Wakhlu AK. Long-term results of anterior sagittal anorectoplasty for the treatment of vestibular fistula. J Pediatr Surg 2009;44:1913-9.  Back to cited text no. 6
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7.
Waheeb SM. The anterior sagittal anorectoplasty technique (ASARP) for the treatment of recto-vestibular fistulae and vestibular anus in children and neonates. Ann Pediatr Surg 2005;1:54-8.  Back to cited text no. 7
    
8.
Dave S, Shi EC. Perineal skin bridge and levator muscle preservation in neutral sagittal anorectoplasty (NSARP) for vestibular fistula. Pediatr Surg Int 2005;21:711-4.  Back to cited text no. 8
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9.
Pratap A, Yadav RP, Shakya VC, Agrawal CS, Singh SN, Sen R. One-stage correction of recto-vestibular fistula by transfistula anorectoplasty (TFARP). World J Surg 2007;31:1894-7.  Back to cited text no. 9
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Wang C, Li L, Liu S, Chen Z, Diao M, Li X, et al. The management of anorectal malformation with congenital vestibular fistula: A single-stage modified anterior sagittal anorectoplasty. Pediatr Surg Int 2015;31:809-14.  Back to cited text no. 10
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11.
Holschneider A, Hutson J, Peña A, Beket E, Chatterjee S, Coran A, et al. Preliminary report on the International Conference for the Development of Standards for the Treatment of Anorectal Malformations. J Pediatr Surg 2005;40:1521-6.  Back to cited text no. 11
    
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Reisner SH, Sivan Y, Nitzan M, Merlob P. Determination of anterior displacement of the anus in newborn infants and children. Pediatrics 1984;73:216-7.  Back to cited text no. 12
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Kuijper CF, Aronson DC. Anterior or posterior sagittal anorectoplasty without colostomy for low-type anorectal malformation: How to get a better outcome? J Pediatr Surg 2010;45:1505-8.  Back to cited text no. 13
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Kumar B, Kandpal DK, Sharma SB, Agrawal LD, Jhamariya VN. Single-stage repair of vestibular and perineal fistulae without colostomy. J Pediatr Surg 2008;43:1848-52.  Back to cited text no. 14
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Menon P, Rao KL. Primary anorectoplasty in females with common anorectal malformations without colostomy. J Pediatr Surg 2007;42:1103-6.  Back to cited text no. 15
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Javid PJ, Barnhart DC, Hirschl RB, Coran AG, Harmon CM. Immediate and long-term results of surgical management of low imperforate anus in girls. J Pediatr Surg 1998;33:198-203.  Back to cited text no. 16
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Liu G, Yuan J, Geng J, Wang C, Li T. The treatment of high and intermediate anorectal malformations: One stage or three procedures? J Pediatr Surg 2004;39:1466-71.  Back to cited text no. 17
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Hamid CH, Holland AJ, Martin HC. Long-term outcome of anorectal malformations: The patient perspective. Pediatr Surg Int 2007;23:97-102.  Back to cited text no. 18
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Kamal JS. Anal transposition trans-sphincteric anorectoplast for recto-vestibular fistula. Saudi J Health Sci 2012;1:89-91.  Back to cited text no. 19
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Shehata SM. Prospective long-term functional and cosmetic results of ASARP versus PASRP in treatment of intermediate anorectal malformations in girls. Pediatr Surg Int 2009;25:863-8.  Back to cited text no. 20
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21.
Holschneider AM, Jesch NK, Stragholz E, Pfrommer W. Surgical methods for anorectal malformations from Rehbein to Peña – Critical assessment of score systems and proposal for a new classification. Eur J Pediatr Surg 2002;12:73-82.  Back to cited text no. 21
[PUBMED]    


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