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Journal of Indian Association of Pediatric Surgeons, Vol. 11, No. 2, April-June, 2006, pp. 85-88 Original Article Early results of the posterior saggital anorectoplasty in the treatment of anorectal malformations in Nigerian children Sowande OA, Adejuyigbe O, Alatise OI, Usang UE Paediatric Surgery Unit, Department of Surgery, Obafemi Awolowo University Teaching Hospital, Ile Ife Code Number: ip06020 Abstract CONTEXT: The ultimate goal of reconstruction in anorectal malformations (ARM) in children, is to create a functional and anatomically aesthetic neoanus.AIMS: To document our experience with the technique of PSARP in ARM and the immediate anatomical and functional outcome SETTINGS AND DESIGN: A retrospective study 39 patients with high and intermediate ARM, managed by the posterior saggital approach, at the Obafemi Awolowo University Teaching Hospital in Ile Ife, Nigeria MATERIALS AND METHODS: Patient's bio-data were collected, as well as, the presentation, investigations, associated malformations, complications, average bowel opening per day and follow up. RESULTS: There were 17 males and 22 females (male: female ratio 1: 1.3). Twenty-three patients (59%) and 15 patients (38.5%), had intermediate and high malformations respectively, while 1(2.5%) had cloaca malformation. The mean age at presentation was 13.9±30.1 month (range 2 day to 15 years), while the mean age at PSARP was 21.6±29.9 months (range 2.5 days to 15 years). 3(7.6%) patients required laparotomy to mobilize the rectal pouch from the abdomen. Mucosal prolapse was the most common significant postoperative problem, occurring in 4(10.3%) patients. The average bowel motion was 3 times daily (range 1 to 8 times per day). CONCLUSIONS: The PSARP is a useful procedure for the correction of ARM, in children in developing countries, even in the absence of muscle stimulators. The immediate anatomical outcome is satisfactory, but functional results are difficult to access because of poor follow up. Keywords: Posterior saggital anorectoplasty, anorectal malformations, Nigerian children Anorectal malformations (ARM) are one of the commonest causes of intestinal obstruction in the newborn. Despite the advances made in its management over the last few decades, affected children continue to be a challenge to the Paediatric surgeons, worldwide. The ultimate goal of reconstruction is to create a functional and anatomically aesthetic neoanus. The posterior sagittal anorectoplasty (PSARP) popularized by deVries and Pena, is regarded as a standard procedure that provides an optimal access to the rectal pouch and any associated fistula and enables proper reconstruction of the muscle sphincter complex around the neo-anus, in children with anorectal malformations.[1],[2] Studies have shown that the functional and anatomic outcome is better than sacro-perineal and sacro-abdomino-perineal operations and the complication rate is low.[3],[4] Also, there is little interference with nervous supply to the urinary system, following the PSARP procedure.[5],[6] In Nigeria, anorectal malformation is the commonest cause of neonatal intestinal obstruction. However, there are few reports on the application of PSARP in the management of anorectal malformations in Africa in general and Nigeria in particular, until recently.[7],[8] The aim of this paper is to document our experience with the technique of PSARP in ARM management and the immediate anatomical and functional outcome in Nigerian children. MATERIALS AND METHODS The case notes of all patients with high and intermediate form of anorectal malformations (wingspread classification), who had the posterior sagittal anorectoplasty at the Obafemi Awolowo University Teaching Hospital between May 1994 and December 2003, were retrospectively reviewed. Information on the patient′s bio-data, presentation, investigations, type of anomaly, associated malformations, complications, average bowel opening per day and follow up were retrieved. Invertogram was done only when necessary, where there are no external perineal fistulae. Children with intermediate or high malformations were preferably given colostomy and a colostogram was performed to determine the nature and location of any fistula where present, prior to repair. All the patients had bowel washout via the stoma and peri-operative antibiotics were given. The operative procedure was as described by Pena et al [Pena and deVries], with some adaptations. The area of maximal sphincter contraction was identified preoperatively and under light anaesthesia, by stimulation with pin prick or digital stimulation. During the operation, the muscle fibres were identified visually and by low current diathermy, as there was no muscle stimulator available. Identified muscle groups are marked with sutures on either side of the posterior sagittal incision. Where the rectal pouch remains too high for mobilization via the perineal wound, the levator muscle and the sphincter complex are reconstructed around a size 24 or 26 Foley′s catheter, pushed as far as possible into the pelvis. The patient is then placed in the lithotomy position. Laparotomy with intra abdominal mobilization of the rectal pouch is done and the previously inserted catheter is now used to guide the rectum to the perineum, where the neo-anus is constructed. Postoperative dilatation was commenced 10-14 days postoperatively, provided wound healing had occurred and continued daily, until the colostomy is closed. The patient is only discharged after the anus can accept the maternal little finger. Thereafter, it is continued in the outpatient basis by the mothers. During the clinic, the mothers are asked about the daily bowel habit, including the presence of soiling and straining. The complications of the procedure, as well as the anatomic and functional outcome in term of bowel motion as obtained from the mothers at follow up clinics, were noted RESULTS Thirty-nine children had the posterior sagittal anorectoplasty (PSARP) for high and intermediate anorectal malformations, during the study period. There were 17 males and 22 females (male: female ration 1: 1.3). Twenty three patients (59%) had intermediate malformations, while 15 (38.5%) had high type malformations and 1(2.5%) had cloaca malformation. The mean age at presentation was 13.9±30.1 month (range 2 day to 15 years). The types of anorectal malformations at presentation, are as shown in [Table - 1]. Two of the patients with high type malformation had flat perineum, suggesting a possibility of incontinence, postoperatively. Gross associated malformations were seen in 7(17.9%) patients,and are as shown in [Table - 2]. One of the patients had suspected VATER′S association. The mean age at PSARP was 21.6±29.9 months (range 2.5 days to 15 years). Of the 39 children who had the posterior sagittal anorectoplasty, 3 required laparotomy to mobilize the rectal pouch from the abdomen, while a vaginoplasty was added in 1 and posterior sagittal anorecto-vagino-urethroplasty (PSAPVUP) was done in the patient with cloaca malformation. Two patients with recto-vestibular fistula had attempted primary PSARP, but required colostomy following wound breakdown and retraction. Repeat PSARP was then performed after 3 months. Significant complications were observed in 12 patients (30.8%). Mucosal prolapse was the most common and occurred in 4 (10.3%) patients, but was only troublesome in only one patient, requiring excision. One patient had a redo PSARP, because of dehiscence and retraction. Other complications are as listed in [Table - 3]. There was one (2.7%) mortality, but not directly related to the procedure, as the child developed postoperative malignant hyperthermia and subsequently died. Follow up following neo-anal reconstruction has been poor, as only 31 patients had follow up ranging from 1 month to 63 months (mean 12 months). Information on bowel habit was available for 33 patients. The average bowel motion was 3 times daily (range 1 to 8 times per day). Only 11 of these 33(33.3%) of the children were above 3 years at the time of surgery or during follow up. 10(91%) were fully content of faeces and gas, while the remaining one had occasional soiling. DISCUSSION The management of infants with ARM continues to be a challenge to paediatric surgeons, world wide. For optimal results, careful preservation of structures and precise anatomical reconstruction is required. As a result of this, there are numerous operations devised for the treatment of these conditions. However, the PSARP approach for the repair of anorectal malformations in children, which was introduced in the early 80s by deVries and Pena, has since become widely accepted, as the procedure of choice in the treatment of anorectal malformations in children.[4],[9] PSARP enables the surgeon to repair the pelvic anatomy under direct vision, utilizing all the existing continence muscles and hence resulting in better faecal continence. With this approach, the entire spectrum of ARM can be repaired. The result of cosmesis is generally regarded as better and the immediate postoperative complications are few.[2],[4] The procedure has been adapted for various other surgical conditions involving the pelvis and perineum, both in children and adults. Despite the world-wide acceptance of this procedure, there appears to be reluctance in the use of PSARP in Nigeria and perhaps the rest of Africa, as there are few reports in the literature of the clinical application of the operation in our environment, until recently.[7],[8] Over a period of 10 years, we have applied this procedure on 39 children with intermediate or high type of anorectal malformations, with good anatomical results. The immediate anatomical appearance of the neo-anus and perineum has been satisfactory. The operation requires a good understanding of the anatomy, good surgical skills and patience, so as to avoid complications. In most centres in the developed world, the operation is carried out in the immediate neonatal period, without the use of colostomy. This is said to give a better functional outcome, probably from the early development of the necessary cortico anal somatosensory integration, which may be lost in those patients who had a diverting colostomy. The absence of neonatal intensive care unit makes early neonatal reconstruction impossible in our centre. Therefore, we tend to give our patient colostomy before surgery. We have also found colostomy to be a live- saving measure, as most of our patients also present with intestinal obstruction and are usually in bad clinical state. In the older children presenting with ARM, we preferentially give colostomy to divert faeces from the operative site, so as to avoid wound complications. Moreover, correct placement of the neoanus within the muscle sphincter requires that the muscle be identified and divided in the midline. This is facilitated by the use of Pena′s muscle stimulator. However, this is not available in our unit. We rely on visual identification of the muscle at operation, as well as low current diathermy stimulation. In most of our patients, this was easy, as most of them were over six months at the time of the surgery. An improperly placed anorectum can send the child to a life of incontinence, or the need for repeated surgery. This luxury is not available, as most of our patients are poor and it takes considerable expenses to complete treatment. This might explain why many of the patients are lost to follow up after the initial colostomy.[10] It is therefore important to get the reconstruction right at the first go. In many series, using a variety of continence scoring system, continence following PSARP ranges from 35 to 78%. [11],[12],[13],[14],[15] Continence is however dependent on a variety of factors, including type of anomaly, absence or presence of the sacrum and the sex of the patient.[15] In our series, the functional outcome is difficult to determine, because of poor clinical follow up and poor communication. Also manometric and other expensive modalities of assessing sphincteric functions, are not available. Our experience based on maternal reports in 11 of the children who had attained the age of 3 years at the time of their surgery or at follow up, suggests that the functional outcome in terms of continence would be good. Moreover, none of the patients we have operated upon, has reported back with intractable faecal incontinence. Many other studies have suggested that the functional outcome after the PSARP is better, than in patients with the sacro-perineal or the abdomino-sacro-perineal pull-through, although the outcome is also influence by the presence of associated anomalies, especially sacral agenesis, which adversely affects the outcome. None of our patient had such anomalies. The complication rate in our centre is 30.8%, which is high, though comparable to 26% quoted by Nakayama et al . This excludes superficial wound infections that healed spontaneously. Most complications following the PSARP are said to be preventable, with attention to operative techniques. There was no major wound infection or dehiscence, most probably because of the presence of proximal colostomy. Rectal prolapse occurred in four of our patients, but only one required excision of the prolapse. This patient had suspected VACTERS association. The overall incidence of significant rectal prolapse following PSARP is low. Patients with higher anorectal malformations, poorer muscle quality and vertebral anomalies, had a greater risk of developing postoperative rectal prolapse.[16] Intra-operative complications like vaginal perforations in girls and urethral injuries in boys are infrequent, but can occur. Hong et al found that 129 of 1003 children had urologic injuries following reconstruction of anorectal malformations. 26 of these children had urethral injuries.[17] Early recognition and immediate surgical closure at surgery, is all that is needed. Two of our patients with intraoperative vaginal perforation did well postoperatively, but the patient with urethral injury died of non associated causes. Repeat PSARP is indicated in situations, where there is major disruption of the neoanus associated with retraction or for faecal incontinence, where the rectum was not placed within the muscle complex, or occasionally to treat recurrent recto urinary fistula.[18],[19] We only had to do repeat PSARP in 2 patients as a result of retraction of the neoanus, with satisfactory outcome. We recorded only one case of Anal Stenosis, which was managed successfully by repeated anal dilatation. Anal stenosis usually follows non compliance with dilatation regimen, post PSARP. It is our practice to instruct the mother to continue with digital anal dilatation at home, because it is impossible to give all the patients Hegar′s dilator to take home. In conclusion, the PSARP is a useful procedure for the correction of ARM in children in developing countries, even in the absence of muscle stimulators. The immediate anatomical outcome is satisfactory but functional results are difficult to access, because of poor follow up and lack of facilities. References
Copyright 2006 - Journal of Indian Association of Pediatric Surgeons The following images related to this document are available:Photo images[ip06020t1.jpg] [ip06020t3.jpg] [ip06020t2.jpg] |
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