During follow-up, none of our patients were subjected to a routine anal dilatation program as recommended by Keily and Pena 9. We only calibrated the anal opening to ensure that there was no stenosis during the follow-up visits to our outpatient clinic. All our patients received a barium enema to assess the rectoanal angle (RAA), an ascending and voiding cystourethrogram (ACUG and VCUG) to exclude presence of a residual urethral diverticulum (RUD), and an MRI to assess rectal placement within the levator ani and external sphincter muscle complex. Functional outcome was determined in patients older than 3 years only using a functional continence evaluation questionnaire 6.
All our living patients (except those requiring a redo-PSARP) received a barium enema in the postoperative period after closure of their colostomies. The RAA was determined by measuring the angle between a line drawn through the central portion of the anal canal and a line drawn parallel to the posterior wall of the rectum. Barium studies were performed on the patient during rest in the lateral decubitus position 10.
The ACUG and VCUG were done to exclude the presence of a posterior urethral diverticulum or a recurrent rectourethral fistula. The catheter was introduced into the bladder, and a dye was injected filling it, then the catheter was gradually withdrawn while injecting the dye to demonstrate whether a fistula or diverticulum was present. The presence of vesicoureteric reflux was also noted 11.
A pelvic MRI was performed on all our living patients (except those requiring a redo-PSARP) to assess the rectal placement within the levator ani and external sphincter muscle complex. Cuts were evaluated at the level of the puborectalis and external sphincter at 3 and 9 o’clock positions for the symmetry of the muscle thickness around the rectum. A score of 0 was given if the muscle around the rectum was symmetrical (Fig. 3), slight asymmetry was given a score of 1, and marked asymmetry was given a score of 2. The worst score was 4, 2 for each muscle level 6,12. All of these radiological investigations were performed during the postoperative period 2–3 months after closure of the colostomy.
Functional outcome was evaluated using a score proposed by Ichijo et al.6 (Table 1). Only patients who were older than 3 years were included. Patients who had tethered cord, had gross sacral agenesis, or had a redo-PSARP were excluded.
Twenty boys were enrolled in our case series. We had a mean sacral ratio of 0.82±0.19. Our study included 11 patients with rectourethral bulbar fistula (RBF), seven with rectourethral prostatic fistula (RPF) one with rectovesical fistula, and 1 with no fistula. Thirteen of our patients showed associated anomalies with two patients showing VACTREL association. The various associated anomalies are shown in Table 2.
The mean age at time of surgery was 236±77 days. Our mean operative time was 152±32 min Fistula closure in all our patients was done using titanium clips, except in one baby who did not have a rectourethral fistula. Regarding intraoperative complications, four patients had a breach of the rectal wall with peritoneal contamination which was managed by suction irrigation and placement of a pelvic drain and postoperative parenteral antibiotics. One patient experienced a diathermy injury to the left ureter which required a resection anastomosis owing to development of ureteric stricture. One patient had a thermal injury to his bowel from using a harmonic scalpel requiring inversion with interrupted seromuscular sutures extracorporeally. One patient experienced significant bleeding requiring a blood transfusion. No conversion to open surgery occurred.
Median hospital stay was 5 days (range: 3–18 days). All our patients were started on oral feeding with 24 h, except the one patient with thermal injury to his bowel where feeding was delayed for 3 days. A self-retaining urinary catheter was placed in all patients for 5 days postoperatively. Two patients experienced a neurogenic bladder which was discovered after catheter removal. The catheter was replaced in both patients, and they were discharged home. The catheter was removed 4 weeks later, and both boys voided normally. One patient developed severe port site infection and complete disruption of his rectocutaneous anastomosis. This patient had chronic kidney disease owing to obstructive uropathy from a neuropathic bladder. He had a vesicostomy created before his LAARP elsewhere. We concluded that infection spread from his bladder owing to the presence of Urinary tract infection causing this complication. This was documented with a urine analysis. This was compounded by his immune-compromised state secondary to his chronic kidney disease. He was managed with regular dressing, debridement and aggressive intravenous antibiotics till the infection cleared. He was discharged on the 18th postoperative day. None of our patients experienced postoperative peritonitis, evisceration at any port site, or ischemia of the pulled-through colon.
One of our enrolled patients died during follow-up (1 year after the procedure) owing to pneumonia and sepsis. None of our patients received regular anal dilatations postoperatively; only calibration was done during their visit to the outpatient clinic. Eight patients experienced variable degrees of mucosal prolapse (40%) that developed within 2 months of the initial LAARP. Seven of these were minor prolapses. All eight patients were readmitted 6–8 weeks later for mucosectomies. None of our patients experienced anal stenosis. The one patient who had an infection at the site of the rectocutaneous anastomosis experienced a complete disruption with retraction of the rectum that required a redo-PSARP. One had partial rectocutaneous anastomotic disruption and was readmitted for a redoanoplasty. One patient was judged to have a misplaced rectum and was also managed with a redo-PSARP. Patients who had redoanoplasties or redo-PSARPs were started on regular anal dilatations 2 weeks postoperatively for 5 months according to the schedule recommended by Keily and Pena 9. This was done by the parents at home.
The mean obtained fecal continence evaluation questionnaire score obtained for 11 eligible patients was 7.09±1.12. Overall, 17/19 patients received a barium enema, revealing a RAA of 107±13°. All 19 available patients received an ACUG and VCUG. Moreover, 7/19 (37%) patients showed a posterior urethral diverticulum; six of these patients had RBF, representing 87% of all patients with RUD. Mean MRI scores obtained were 0.76±0.51.
The 1980s is considered to be the starting of the golden era for surgical management of ARMs as it witnessed the birth of the PSARP 2. However, the rate of postoperative motility disorders in the form of constipation and associated megarectum started to climb. Georgeson et al.1 proposed LAARP as an alternative in the year 2000.
Using LAARP to manage rectovesical or high rectoprostatic fistulas is logical. However, implementing it to treat ARMs with RBFs or in absence of a fistula remains controversial. Koga et al.12 performed a comparison between LAARP and PSARP in RBFs only and concluded that LAARP provided comparable results to PSARP in such babies with lower risk of wound infection but a higher risk for rectal mucosal prolapse. We wanted to investigate this further and decided to include patients with RBF in our study.
A major point of criticism in our study is that we did not perform regular anal dilatation after the initial LAARP. We only calibrated the anal opening with Hegar dilators during follow-up visits to our outpatient clinic. An anal dilatation regimen was reserved for patients who required a redoanoplasty only. This was because our first case experienced rectal prolapse requiring a mucosectomy developed anal stenosis during follow-up. He responded well to anal dilatation which was performed at home. We decided to employ a dilatation program there on for other patients who would require a redoanoplasty. This is not the standard practice reported by many authors who advocate anal dilatation postoperatively to avoid anal stenosis 2–4,11. None of our patients who had LAARP only without a redo-operation experienced anal stenosis in spite the absence of regular anal dilatation.
The most recurring postoperative complication we observed was rectal mucosal prolapse. Overall, 40% of the patients in our study experienced this. This is one of the most frequently recurring complications following LAARP. A prolapse rate of up to 52% has been reported in publications 13. In our study, this could be explained by the fact that we did not place anchoring sutures to fix the rectum to the presacral fascia as recommended by Georgeson 5. However, this is a matter for debate. Jung et al.13 and Ruggeri et al.14 placed more emphasis on decreasing the length of the rectal dissection to reduce the risk of postoperative prolapse whereas authors such as Leung et al.15 stressed on the value of the rectopexy stitches in prevention of prolapse. A combination of both of these may be successful in greatly reducing our prolapse rates in the future.
Our second most recurring problem was RUD. Overall, 37% of our patients developed varying degrees of this. This occurred much more frequently with patients with RBF than with the other types of ARM in our study. This could be explained by the long common wall between the rectum and the urethra and the upward angulation of the fistula as it terminates into the bulbous urethra, which obscures the view when using the laparoscope. This makes total resection of the fistula a challenge. Moreover, there is an inherent risk of damage to the urethra coupled with our early inexperience with deep pelvic dissections using laparoscopy which made us shy away from taking our dissection too far. This is what may explain our high incidence of post-LAARP RUD, which was the same explanation offered by Jung et al.13.
To overcome this problem, several solutions were recommended by many authors. Some suggested completely abolishing the use of LAARP in patients with RBF and restricting it to ARM with RPFs or rectovesical fistulas 16. Other authors proposed modifying the technique to deal with RBFs and avoid the development of a RUD 10,11,14,17,19. Till now, none of our patients who developed a RUD showed any symptoms or were subjected to surgical resection.
The results obtained when calculating the RAA in our study group reflected our ability to attain good rectal placement within the levator ani and puborectalis sling. We coupled this with MRI studies of the pelvis. Our MRI scoring system was based on that reported by Ichijo et al.6 and Koga et al.10. Scores obtained using both these methods reflected our ability to place the rectum within the confines of the muscle sphincter with a high degree of accuracy. We should state however that interpretation of the MRI may be subjective affecting the results obtained 6.
To evaluate the functional outcome, we adopted the fecal continence evaluation questionnaire (FEQ or CEQ) 6. We chose this as it was most suited for our study. This is because we wanted to offer a functional evaluation of the technique, but most of our patients would not have passed the age of 4 years, which is required to implement scores such as the Kelly continence score 3,14, the Krinkenbeck score 10, and the Rintala questionnaire 14,18. Our results reflected a good outcome for LAARP regarding continence; however, annual measurements to assess progression are required. We are also thinking of applying other scores to evaluate functional outcome when our patients get older in the future.
LAARP offers a great alternative to manage intermediate and high ARMs in boys; however, this is not without its problems. Rectal mucosal prolapse and RUD remain the most frequently recurring complications following LAARP. Methods for prevention of these complications must be more objectively assessed in the future.
Conflicts of interest
There are no conflicts of interest.
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