Pediatr Surg Int (2007) 23:1191–1197 DOI 10.1007/s00383-007-2019-2
ORIGINAL ARTICLE
Anterior sagittal anorectoplasty for anovestibular fistula Sanjay Kulshrestha Æ Meeta Kulshrestha Æ Balbir Singh Æ Barun Sarkar Æ Mukesh Chandra Æ A. N. Gangopadhyay
Accepted: 3 September 2007 / Published online: 27 September 2007 Ó Springer-Verlag 2007
Abstract Anterior sagittal anorectoplasty (ASARP) was used for the definitive correction in 107 cases of anovestibular fistula (AVF) between 1996 and 2005. These cases were subjected to three different types of treatment regimes during the same period. Majority of the cases (78) were operated in one stage where postoperatively an early oral feed was started (A). Cases were discharged in 2–4 days. In the second group (B), there were ten cases who were also operated in one stage but with prolonged fasting of 9– 10 days postoperatively. Nineteen cases (C) were operated under cover of colostomy during the same period. In the immediate postoperative period, among the group A, one case had a major wound disruption requiring a colostomy and a redo surgery. Three cases had subcutaneous leak. In seven cases there was premature dehiscence of mucocutaneous or skin sutures. In groups B and C, there were no significant complications in the immediate postoperative period. In the follow-up period, out of 107 cases, 63 (58.8%) had constipation at the end of 3 months. However, at the end of one year, only 24.3% (26 cases) cases had constipation. Regarding fecal continence, 86 cases (90.5%) were totally continent. Seven had history of occasional soiling and in two cases, soiling was more frequent. As far as repair or correction of AVF or vestibular anus is concerned, we feel that anterior sagittal approach is more suitable as it requires less pelvic dissection. Separation of posterior vaginal wall from rectum, which is considered, is S. Kulshrestha M. Kulshrestha B. Singh B. Sarkar M. Chandra A. N. Gangopadhyay Division of Pediatric Surgery, Sarkar Hospital for Women and Children, Agra 282002, India S. Kulshrestha (&) 1/188-C, Delhi Gate, Gulab Rai Marg, Agra 282002, India e-mail:
[email protected]
the most important step of the operation, takes place under direct vision. We also feel that AVF can be repaired in one stage with an early postoperative oral feed, provided we are meticulous in pre and postoperative bowel management. It reduces hospital stay and the cost of treatment. This provides a good option to cases who are not able to afford prolonged hospitalization (fasting) or are not willing for a colostomy. Keywords Anterior sagittal anorectoplasty Vestibular anus Anovestibular fistula Rectovestibular fistula Anal transposition Anorectal malformations
Introduction Anovestibular fistula (AVF) or vestibular anus is the commonest malformation amongst female anorectal malformations (ARM), which a pediatric surgeon comes across. More importantly, the gravity of its surgical correction is frequently underestimated. Beside this, a significantly high incidence of postoperative constipation as compared to other ARM makes AVF a unique anomaly. However, as compared to male anomalies or other more complex high female anomalies, this has received less attention than it probably deserves. In the neonatal period, a cutback procedure is recommended for AVF and thereafter, various methods are available for its definitive correction. These are Y–V plasty [1], perineal anal transposition (Potts) [2], X plasty [3] and X–Z plasty [4]. Inadequate exposure, incomplete rectovaginal separation with subsequent tendency of new anus to move forward, and blind placement of anorectum in the sphincter complex are the main disadvantages making
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these procedures less popular. Better exposure and precise placement of the anal canal within the external sphincter complex have made the posterior and anterior sagittal approaches more popular and established for the correction of AVF. Pena [5] recommended a posterior sagittal approach for this anomaly and later on this approach has also been used by others [6–8]. Anterior sagittal approach that utilizes the same basic principles as that of the posterior one was first reported by Okada [9] and later on by Wakhlu [10] and Aziz [11]. Differences in opinion exist regarding the need for a protective colostomy in AVF. Repair under cover of a colostomy is considered the safest option for these cases [12, 13]. On the other hand, reports of one stage correction of vestibular anus are also available [7–11, 14]. Even amongst those who recommend a one stage repair, the opinion differs in whether an early postoperative feed should be given or not. Okada [9] and Sanchez [6] have recommended that prolonged fasting for 2 weeks is necessary for the proper healing of the wound. Others [7, 8, 10, 11, 14] feel an absolute fasting is not necessary for these cases. Herewith, we are presenting our experience of operating AVF by anterior sagittal anorectoplasty (ASARP) through different regimes available.
Pediatr Surg Int (2007) 23:1191–1197 Table 1 Relative incidence of various ARM in females Anomaly
No. of cases (%)
Perineal fistula and ectopic perineal anus
61 (22.6)
Anovestibular fistula
107 (39.6)
Rectovestibular fistula Rectovaginal (low and high)
8 (2.96) 19 (7.1)
Pouch colon (Colovaginal fistula)
10 (3.7)
Cloaca (included 5 pouch colon)
33 (12.2)
Congenital ‘‘H’’ type fistula Low (Anovestibular or perineal canal)
20 (7.4) 12 (4.44)
Intermediate (rectovestibular)
5 (1.85)
High (rectovaginal)
2 (0.74)
Imperforate anus with absent vagina (A normal size rectum terminated at vestibule looking like vagina with no actual vagina)
5 (1.85)
Pure atresia (anorectal agenesis without fistula)
3 (1.1)
Rectal atresia with normal anus
1 (0.37)
Imperforate anus with double abnormal termination of fistula (Two anterior openings: one high vaginal and one vestibular)
1 (0.37)
Materials and methods
Rectal atresia (normal anus) with rectovestibular fistula
1 (0.37)
Two hundred and seventy cases of female ARM were seen in our hospital from June1996 to February 2005. Table 1 shows the distribution of various female ARM. One hundred and seven cases (39.6%) were found to be having the AVF. Twenty-one cases (19.6%) had an associated anomaly (Table 2). Those who presented in the neonatal period were operated at around 3 months of age. The age of the patients at operation ranged from 3 months to 17 years (Table 3). The median age at operation was 10.5 months. Out of a total of 107 cases, 45 (42%) presented within 3 months of age and amongst them, 23 cases had an anal stenosis requiring a cut back operation at the time of presentation. Rest of the cases could be managed by dilatation alone. Out of the 62 cases that presented after 3 months, 16 had anal stenosis at the time of presentation. Eleven of these cases had a cutback operation while in five cases, who had severe constipation, the definitive procedure was planned under cover of a colostomy. After this primary management, we recommended a definitive procedure in all these cases. All these 107 cases of AVF were operated by ASARP. The main criteria for selection of AVF were: (1) the fistulous opening is below hymen in the vestibule and is surrounded by a pink mucosa all around; (2) the lumen of intestinal tract remains normal right up to the vestibular opening. All these 107 cases were divided
Total
270 cases
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The incidence of low female ARMs was 66.6% (i.e. ectopic perineal anus, anovestibular fistula and low type congenital ‘‘H’’ fistula or perineal canal)
into three different groups according to the treatment regimes used for their treatment: (A) One stage repair with early postoperative feeding–– 78 cases (B) One stage repair with prolonged fasting postoperatively––10 cases (C) Repair under cover of a colostomy––19 cases. The indications for the colostomy were: (1) after explaining the risk of one stage repair, parents opted for operation under cover of colostomy (14 cases). (2) Those cases who presented with anal stenosis with severe constipation (5 cases). In these 19 cases, a colostomy was done in the same sitting just preceding the definitive operation. For group B (prolonged fasting), we selected those patients who were above 3 years of age where a good cooperation was expected from the patients. Though initially we selected 12 patients for group B, during the postoperative period in two cases, an early feed had to be resumed on the parent’s insistence. Although the basic steps of the operation were
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Table 2 Associated anomalies (amongst 107 cases) Type of anomaly
No. of cases
Spina bifida
2
Tracheoesophageal fistula
2
Duodenal atresia
1
Cardiovascular anomalies
3
Limbs (a) Congenital dislocation of hip
1
(b) Congenital talipes equinovarus
2
(c) Congenital syndactyly
2
Ureteropelvic junction obstruction
1
Vesicoureteral reflux
2
Cleft lip Double vagina
2 1
Partial sacral agenesis
2
Total
21 (19.6%)
Table 3 Table showing age at operation Age group
No. of cases
3–6 months
16
6–9 months
28
9–12 months 1–2 years
20 18
2–3 years
11
3–4 years
7
Above 4 years (up to 17 years)
7
Total
107
The median age at operation was 10.5 months
the same in all these three groups, there were some differences in the pre and postoperative management. Mechanical bowel cleansing was achieved in all cases of groups A and B by oral osmotic agents [15]. In 77 cases, we used polyethylene glycol with electrolytes (pack of 137 g dissolved in 2 liters and this solution was given in the dose of 40–50 ml/kg). In the initial 11 cases we used oral mannitol (10%) at a dose of 8–10 ml/kg. Osmotic agents were administered 8–10 h before operation. After oral osmotic agents, only plain water was allowed which was stopped 4 h before the operation. Preoperative enema was used only if effluent was not clear after oral osmotic agents. The important steps of operative technique have been mentioned along with figure legends (Fig. 1). During postoperative period for group A cases, a liquid diet was started from second day and continued for 8–10 days. Thereafter, a semisolid diet was given. Oral laxatives were started from second day with an aim to achieve a smooth and effortless defecation without any straining. Regarding the wound care, the parents were explained to clean the
wound by povidone iodine lotion with application of antibiotic ointment 3–4 times a day. IV fluids were stopped in 2–3 days. Total hospital stay was from 2 to 4 days. For group B cases, only plain water was allowed. All the patients were kept on IV fluids and partial parenteral nutrition for 9–10 days. Thereafter, a liquid and semisolid diet was allowed. All cases were subjected to anal dilatation after 3 weeks. A daily dilatation was advised for first 3 months and then twice a week for the next 3 months.
Results In the immediate postoperative period amongst the group A patients, one case had a major wound disruption on the sixth day for which a sigmoid colostomy was done. A redo perineal repair was performed by the anterior approach after 5 months and 2 months later, the colostomy was closed. Three cases had subcutaneous leak with the history that in addition to passing stool through anal canal, a small amount was also coming out through the anterior end of the incision (i.e. formation of acquired perineal canal). In all the three cases, the skin stitches were laid open and the wound was allowed to heal, i.e. converting them to a perineal groove. Three months later, this groove was repaired in two cases while in the third one, the parents were satisfied with the residual scar. In seven cases, there was premature dehiscence of mucocutaneous or skin sutures without any significant residual scar after healing by secondary intention. There were no significant complications in the immediate postoperative period in groups B and C. All these patients were followed up in a prospective manner for at least 5 years (every month for first 3 months, every 3 months for the first year, then six monthly for next 4 years). Assessments were made for: (1) number of voluntary bowel movements in a day, (2) history of constipation if any and frequency of requirement for laxatives and/or enema, (3) about fecal continence or soiling accidents. Depending on the status of continence, cases were divided into three groups: (A) totally continent without any history of staining under stress, (B) occasional soiling (once or twice in a week) or soiling under stress, (C) more frequent or almost daily soiling or soiling even without any stress. Out of 107 cases, 63 (58.8%) had constipation at the end of 3 months which required laxatives. In eight of these cases, in addition to laxatives, an enema (once or twice a week) was also required. However, at the end of one year, only 26 cases (24.3%) had constipation. Five out of these 26 cases had severe constipation requiring daily laxatives and twice a week enema while the remaining 21 cases had moderate constipation requiring laxatives on alternate day basis with occasional enema.
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Fig. 1 Operative steps of ASARP for anovestibular fistula. a Midline incision extending from the fistula up to the center of proposed new anus. The incision is deepened by dividing perineal muscles and anterior fibers of external sphincter complex. b Exposed posterior wall of rectum. Posterior to rectum there is a well-defined plane where a blunt dissection can be performed. A complete posterior dissection is not done at this stage. After mobilizing 3–4 cm of rectum, we move on to lateral dissection. c Anterior dissection started. A sharp dissection is needed to enter the plane between posterior vaginal wall and rectum. It requires extreme patience, good illumination and proper assistance. d Anterior dissection continued. It is done up to the level of cervix. In author’s experience the firm union between posterior vaginal wall and rectum is present right up to the
level of cervix (unlike male rectobulbar fistula where after 2–3 cm dissection above fistula, we get a loose plane). e Rectal mobilization completed. The deeper anterior dissection is done simultaneously with the remaining posterior and lateral dissections. During posterior dissection, the levator ani muscle does not require division in vestibular anus. f Perineal reconstruction started. The mobilized anorectum is placed within the limits of external sphincter complex and starting from the deepest tissue first it is approximated in layers by interrupted suitably sized vicryl sutures. g Perineal reconstruction continued. The rectum is fixed to posterior most stitch of each row and a few stitches are also used circumferentially to fix lateral and posterior walls of rectum to the surrounding muscles. h After complete reconstruction
Those cases that required occasional laxatives (once a week) were not included in the constipation group or were considered cured as far as constipation was concerned. Fecal continence was assessed in 95 cases (cases of spina bifida, sacral anomaly and girls having age less than 3 years were excluded). Eighty-six cases (90.5%) were totally continent (group A), seven had history of occasional soiling (group B) and in two cases soiling was more embarrassing as occurring more frequently (group C). One of these two cases of group C was the same who had a major wound disruption. The problem of constipation improved with time. However, as far as incontinence is concerned, we did not find any significant improvement with time.
Discussion
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AVF and perineal fistula are the two important low ARM in females. As per classification, only a change of few millimeters in the termination of fistula posteriorly, makes an AVF a perineal one. However, as far as management of these two low anomalies is concerned, the approach or treatment of AVF is entirely different from that of perineal fistula. A firm union between posterior vaginal wall and the rectum in AVF fistula requires much more technical skill making definitive correction more difficult as compared to perineal one. The conventional cut back operation may probably be regarded as a definitive procedure in perineal fistula but certainly cannot be regarded
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as a definitive procedure in AVF. In female ARM, we have two basic aims at operation viz. adequate separation of vagina from rectum and adequate downward mobilization of rectum to perform a tension-free anastomosis with skin. However, if we consider the AVF, we see that terminal gut is of normal caliber right up to its termination. So as far as mobilization of rectum is concerned, just separation of rectum all-round gives enough length and we hardly need any extra dissection for the purpose of downward descent (that is why we name this operation as anal transfer or transposition rather than a pull through). Undoubtedly the posterior approach gives a better view for the posterior dissection of rectum. However, we also know that posterior dissection in these cases is much easier or simpler and does not require much exposure. We feel that the wide exposure obtained in posterior sagittal anorectoplasty (PSARP) by dividing the perineum into two halves is probably more than what we really need for AVF. We feel that posterior sagittal approach is more useful in cases of higher female anomalies where we need more extensive downward mobilization of the rectum.
Upper limit of dissection To prevent the mobilized anorectum from receding inside and forward, Pena [12] has advocated anterior dissection up to a point where rectum and vagina separate and have full thickness walls. Okada [9] feels mobilization of 4– 5 cm length of rectal tube is adequate. According to Zanotti [16], dissection is required up to the body of uterus. Considering normal anatomy of perineum, we see that genital and intestinal tracts start separating from each other at the level of cervix. We feel dissection up to level of the cervix or posterior fornix is adequate to restore normal anatomy.
Are PSARP and ASARP the same operation in two different positions? The answer is no. There are two basic differences between the two. (1) In the anterior approach the incision is much smaller and stops exactly at the midpoint of the proposed new anus while in the posterior approach, it extends more backwards up to the coccyx. Anterior approach requires division of only anterior fibers of external sphincter complex while posterior fibers remain intact. Thus the amount of tissue dissection in posterior approach is more putting a larger area at risk in case if infection occurs. A modified or mini PSARP [17], where the incision is same as that of ASARP, has also been described for the simpler female
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ARM (perineal or fourchette fistula). To our knowledge, no reports are available that indicate whether this mini PSARP also suits to vestibular fistula. (2) In the anterior approach, anterior dissection, i.e. separation of vagina and rectum, takes place under direct vision while in posterior approach this dissection is blind.
Need for a protective colostomy As far as perineal fistula is concerned, there is a consensus that it can be performed without a colostomy. Similarly in rectovaginal fistula or higher anomalies, a colostomy is strongly recommended. However, differences of opinion exist for the need of colostomy in AVF. Pena [12] and Hienen [13] feel the need of colostomy in AVF. They have very valid reasons for saying this. Firstly, it is an anomaly where satisfactory results can be achieved to the tune of 90–95% and any major complication would mean three additional operations, i.e. colostomy in the immediate postoperative period followed by a redo repair and finally a colostomy closure. In spite of this, same results cannot be achieved in subsequent redo surgery. Secondly, the amount of dissection in AVF is almost the same as that of high lesions thereby requiring a protective colostomy. Those who recommend a one stage repair feel that there are some definite advantages of not doing a colostomy: (1) total correction is accomplished in one instead of three stages. This avoids repeated hospitalizations and total cost of treatment. (2) The colostomy formation is not a minor operation. Reports [18, 19] have shown a significant incidence of major complications in relation to colostomy formation. Similarly, colostomy closure is still not a very safe operation and carries significant morbidity [20] and mortality [21] (0–5% among various series). We have performed 88 such cases in one stage with one major disruption. We feel it is important to mention that we have started doing one stage repair only after doing a considerable number of cases under cover of colostomy. Here, it is not our contention that ASARP is better or safer than other methods available if one does not plan a protective colostomy. Instead, we feel the decision for not doing a colostomy should only be made when a surgeon is well experienced and realizes importance of meticulous pre and postoperative bowel management. Even if the surgeon is well experienced, it does not mean that he would necessarily be able to do every case which he comes across without a colostomy. One should not hesitate to do colostomy when parents opt for safer mode of management or patient presents with severe anal stenosis or if rectal perforation occurs during operation.
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Early vs. late postoperative oral feeds after one stage repair One stage repair with prolonged fasting and TPN has been recommended by Okada [9] and Sanchez [6]. We have used this in ten cases with no significant complications. However, it requires prolonged hospitalization where facilities for TPN are available. Besides the poor tolerance for fasting amongst patients, it also increases the cost of treatment. Secondly, keeping patient fasting does not necessarily mean that they will definitely not pass stool for the desired next 8–10 days. If somehow they pass stool at the end of week for the first time, it becomes hard and can harm the suture line.
About constipation As compared to other ARM, the reported incidence of postoperative constipation in AVF is significantly high (up to 50–60%) [13, 22, 23]. In our series, the incidence of constipation was 58.8% in early postoperative period whereas at the end of 1 year, only 24.3% had constipation. We do not know the exact cause for this. The fact that constipation is very uncommon after a cut back operation indicates that it is probably of neurogenic origin occurring due to extensive dissection around the rectum. The other cause could be congenital deficiency or damage to muscle layer on lower anterior wall of rectum that may affect peristalsis. We routinely apply a few stitches in this area to approximate full thickness rectal muscle layer. We also advocate putting a suitably sized anal dilator inside the anorectum before applying the last few posterior most stitches in muscle complex to prevent any tight closure around anal canal. Regarding the management, these cases probably require stool softeners or laxatives for longer duration. A careful history anticipating constipation in every case is necessary to detect and manage it in time. Any patient who was continent after operation but subsequently develops a fecal incontinence should always be looked for fecal impaction causing spurious or overflow incontinence.
Conclusions Amongst the available methods for the definitive correction of the AVF, we feel anterior sagittal approach is more suitable. With adequate pre and postoperative bowel management, satisfactory healing can be achieved after repair of AVF fistula in one stage with an early postoperative oral feed. However, this regime is not free of local complications so it should be followed only after properly
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explaining to the parents about all available regimes with their advantages and disadvantages and with a caution that a colostomy may be required in the immediate postoperative period.
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