RECONSTRUCTIVE SURGERY Appendicovesicostomy as an Alternative Procedure for Patients with Complex Urethral Distraction Defects Amir Reza Abedi1, Saleh Ghiasy2*, Morteza Fallah-karkan3, Seyyed Ali Hojjati2, Jalil Hosseini4 Purpose: Surgical repair of post-traumatic complex urethral stricture poses a major challenge to urologists. Here, we report six patients with irreparable urethral strictures who were successfully treated by using the appendix as conduit for urinary diversion. Materials and Methods: Six patients who had underwent urinary diversion using an appendix during 2015 to 2019 were included in our study. All patients had a history of one or more failed attempts of urethral reconstruction in the past. Mean follow-up for patients was 29 months. Continency was defined as being completely dry for at least 3 hours. Results: Mean age of patients was 40.1 years old (range: 20-70 years). Intermittent catheterization through the conduit was easily performed for every patient without any stomal stenosis. Mild stomal incontinence only oc- curred in one case which was resolved after a few months. All patients were continent during day and night. Conclusion: Based on the results of our study, Mitrofanoff’s technique is a valuable procedure for managing pa- tients with serious complicated urethral strictures who cannot be treated with common standard approaches. Keywords: appendix diversion; Mitrofanoff’s appendicovesicostomy; urethral stricture; urinary diversion INTRODUCTION Urethral stricture and posterior urethral defects are an important clinical problem in male patients(1,2). Road traffic accidents, iatrogenic injuries, and inflam- matory disorders are common causes of urethral stric- tures. In previous studies, the incidence of posterior urethral stricture after pelvic fracture was predicted to be %5-10(3,4). The surgical management of urethral stenosis varies based on etiology, position, length, and thickness of the lesion in addition to the extent of fibro- sis involving the surrounding tissues(5,6). Treatment of stenosis of the bulbar part of the urethra includes exci- sion and end-to-end urethroplasty or a short patch onlay substitution anastomosis(7,8). However, in some patients the urethral defect is so long that it cannot be managed with extensive releasing of urethra from the surround- ing fibrosis, inferior pubectomy, and even re-routing maneuvers(9,10). Various approach have been used to overcome this problem depending on the location and length of the stenosis including oral mucosa graft, en- terourethroplasty, and the combination of dorsal graft with ventral penile flap . However, many complications have been related to these techniques(11-13). In patients with severe and complicated urethral injury, salvage procedures such as perineostomy or suprapubic tube could be performed(14,15). Patients with a history of past surgical procedures, stenosis longer than 3 cm, accom- panying perineal and GI fistulas, presence of diverticu- litis adjacent to the duct, and a non-competent bladder 1Department Of Urology, Shohada-e-Tajrish Hospital, Shahid Beheshti University Of Medical Sciences, Tehran, Iran. 2Men’s Health and Reproductive Health Research Center, Shahid Beheshti Medical Science University, Tehran, Iran. 3Laser Application in Medical Science Research Center, Shahid Beheshti University of Medical Sciences, Tehran, Iran. 4Reconstructive Urology Department, Shohada e Tajrish hospital, Shahid Beheshti Medical Science University, Tehran, Iran. *Correspondence: Men’s Health and Reproductive Health Research Center, Shahid Beheshti Medical Science University, Tehran, Iran. Telfax +98 2122712234, Mob +98 9128198037. Email;Saleh.ghiasy@sbmu.ac.ir. Received September 2019 & Accepted April 2020 neck are defined as complex cases and are not suitable candidates for urethroplasty(13,16). Although the design of a concealed and easily catheterizable stoma in cases with unreconstructable urethral disease was considered a good practical method, the clinical management of these patients still remains to be a dilemma(17). In 1980, Mitrofanoff introduced an alternative procedure for continent diversion in which one end of the appendix was brought to the skin surface as a catheterizable sto- ma and the other end was tunneled into the bladder wall (17-20). Here, we have reported our experience with this surgical procedure in terms of safety and efficacy. MATERIALS AND METHODS Patient Inclusion Five male and one female patient aged 20 to 70 years old (mean age= 40.1) who had underwent urinary di- version using the appendix during 2015 to 2019 at Sho- hada-E-Tajrish Hospital, Tehran, Iran were included in the study. All patients had a history of one or more pre- viously failed surgical attempts of urethral reconstruc- tion and had an long urethral defect involving different anatomic segments of the urethra, or were at risk of uri- nary incontinence after urethroplasty of membranous urethra because of insufficient proximal sphincteric mechanism or patient’s denial to undergo surgery (Fig- ure 1). Due to the reasons mentioned above, patients became candidates of Mitrofanoff urinary diversion. Prior to enrollment, male patients were informed that Urology Journal/Vol 17 No. 4/ July-August 2020/ pp. 386-390. [DOI: 10.22037/uj.v0i0.5592 ] Vol 17 No 04 July-August 2020 387 they would need ART (assistance reproductive technol- ogy) in case of plans for paternity in the future and then informed consent were obtained from all patients. This study was approved by the Ethics Committee of Shoha- da- E-Tajrish hospital. Surgical Technique A lower midline incision was performed to allow si- multaneous access to the bladder, ileocecal junction, and the appendix. After locating the appendix, it was cut separate from the cecum while preserving its mes- entery. Then, an opening was created as the blind end and washed. After passing a 14F catheter down the ap- pendix to check for patency, it was implanted into the bladder through a submucosal tunnel of at least 4 cm length to achieve an anti-reflux effect (Figure 2). Dur- ing surgery, the appendix was dilated with a 14F cath- eter and the catheter was left in-situ for three weeks. The appendix was then secured with absorbable sutures to the bladder muscle and mucosa. The stomal site was prepared based on pre-operative counselling for site se- lection and the stoma was placed at a level proximal to the bladder so that gravity would assist in achieving continence (Figure 3). Also, a cystostomy tube was in- serted for all cases to increase safety measures. Patients were usually discharged 3-5 days after surgery, as soon as they could tolerate solid food. After about 3 weeks, the Mitrofanoff or pouch catheter was removed and the supra pubic catheter was left clamped-off. The patient was taught how to catheterize his pouch/Mitro- fanoff (clean intermittent catheterization) every 3 hours by using a 12 or 14F Nelaton catheter. Occurrence of urinary leakage throughout the period was considered as the patient being incontinent. As for the night, a cath- eter was inserted and secured in place to allow for free urine drainage. If there was no difficulty in catheteriza- tion, the suprapubic catheter was removed after 3 days. After that, cases were frequently followed-up at 3, 6, 18 and 24 months, with special consideration given to patients having difficulties with catheterization and in- continence. Follow-up plan included: stoma evaluation, upper urinary tract ultrasonography, measurement of post-catheterization urine residue, serum creatinine lev- el, and catheter size. Table 1. Patients’ characteristics and procedure outcome. No Age (Year) Defect Length (cm) Follow up, month Cause of Injury Previous surgical Reason for appendix Outcome intervention diversion candidacy 1 70 6 24 Long urethral stenosis One attempt of Urinary incontinency, No residual, post radical prostatectomy cystolithotomy, several long urethral defect No stenosis failed attempts of urethral (from bladder neck to dilation membranous part) 2 36 7 27 Pelvic fracture due to Laparotomy cystography History of twice failed No residual, entrapment under rubble and cystostomy, twice end urethroplasty, No stenosis to end urethroplasty, long urethral defect several urethral dilation attempts,One attempt of stent insertion 3 45 8 36 Pelvic fracture due to Laparotomy and Long posterior No residual, motor vehicle injury cystostomy, Non- urethral defect No stenosis competent bladder neck, internal urethrotomy, pubectomy, failed end to end urethroplasty 4 41 10 30 Fournier gangrene Extensive debridement, History of two failed No residual, several plastic surgeries attempts of urethroplasty No stenosis for scrotal and penile defect 5 60 6 24 Pelvic fracture due Cystostomy insertion, History of twice failed No residual, to motor vehicle injury twice failed urethroplasty,, urethroplasty, incontinency No stenosis orthopedic surgery and long urethral defect 6 20 3 33 Pelvic fracture due to Laparotomy, history of History of once failed No residual, motor vehicle injury once failed urethroplasty, urethroplasty, No stenosis in childhood bladder neck closure risk of incontinence and cystostomy Figure 1. Contrast imaging of all of the included patients. Appendicovesicostomy for urethral distraction-Abedi et al. RESULTS Etiology of urethral defect in our cases included pelvic fracture, post radical prostatectomy urethral stenosis, and necrotic perineal infection (Table 1). The time gap between trauma and Mitrofanoff’s procedure ranged from 18 to 120 months (mean ± SD= 49.3 ± 37.2). Pa- tients’ characteristics and procedure outcome are pre- sented in Table 1. Sonographic evaluation of upper urinary tract during follow-up did not reveal any pathologic findings. Mean serum creatinine level before surgery was 1.2 mg/dL. Mean surgical time was 2 hours (range= 1-3). Average predicted blood loss was around 150 cc (ranged 50 to 600). There was no need for blood transfusion or adja- cent organ injury. All cases were discharged 3-5 days after surgery. Follow-up duration ranged from 24 to 36 months (mean= 29). Post-operative complica¬tions consisting of dehiscence, wound infection, hematoma, necrosis, or perforations of the appendix tube were not detected in any cases during the fol¬low-up period. Catheter size of patients ranged from 12 to 14F. In five of the patients, catheterization was easily performed through the con- duit every 2 hours. Over time, the pouch was expanded to hold more urine and the patient needed to catheter- ize every four to six hours. The only patient who could not easily catheterize underwent flexible cystoscopy and dilation with a 14F catheter. None of the patients had stomal stenosis during the follow-up period. Mild stomal incontinence occurred in only one case who be- came continent after a few months. DISCUSSION The potentiality of the appendix to be used as a con- cealed stoma capable of catheterization was discovered in 1980 by Mitrofanoff(21) in an attempt to achieve uri- nary continence and maintenance of a low-pressure uri- nary storage reservoir(17). Later variations of this tech- nique were developed such as the Monti technique in which a short part of the ileum was used according to the same principle(13,22). The benefit of using the appendix instead of an ileum segment is that intestinal anastomosis is not required in appendicular diversion, thereby the risk of intes- tinal anastomotic leakage is reduced. Also post-oper- ative fasting period is minimized. On the other hand, the physiologic function of the appendix is unknown in adults; therefore, the removal of appendix does not bring a serious harm to the body and does not lead to any impairment in the body’s function. Another disad- vantage of the ileum compared to the appendix is the need for tapering and tabularization which increases the likelihood of urinary leakage. Finally, the most im- portant advantage of using an appendix is shortening of operative time since time-consuming procedures such as ileum-ileum anastomosis and tabularizations are not necessary. The reasons for deciding to create a Mitrofanoff stoma are irreparable loss of the urethra, continence problems, neurogenic bladder with incontinence, unreconstructa- ble bladder (e.g. exstrophy), unreconstructable urethral disease, and congenital anomalies like urogenital sinus (23). The Mitrofanoff principle can also be performed in combination with a bladder augmentation technique.(24) Appendicular diversion can be used in cases who have complicated urethral trauma after accidents(25). The benefits of appendix diversion include maintaining complete continence; easy catheterization; excellent body image; and rarity of post-surgical complications such as dermatitis and urinary tract infection(26). Re- garding the length of appendix ,the cutaneous stoma can be placed in the umbilicus or the lower right abdominal quadrant(27). Yang et al.(28) demonstrated that the sub- mucosal tunnel and abdominal wall muscles are critical factors in the success rate of continence. However, like any other surgery, the Mitrofanoff pro- cedure is associated with some complications such as leakage from the stoma and non-catheterizable channel. Recent reports showed an overall complication rate of Figure 2. Passing a 14F catheter down the isolated appendix to check for patency. Figure 3. The stoma was created at a level relative to the bladder so that gravity would assist in achieving continence. Figure 4. Urethral stricture shown in retrograde urethrogram (RUG) Appendicovesicostomy for urethral distraction-Abedi et al. Reconstructive Surgery 388 Vol 17 No 04 July-August 2020 389 6.2%(18). The incidence of stomal stenosis was 10-23%, incontinence 2-7%(29,30) and stoma revision was required in 16-20% of cases (29,30). Adherence to the technique which provides ease of catheterization intraoperative- ly, wide reflection of the cecum to preserve vascularity, and fixation of the bladder to the anterior abdominal wall guarantees a durable achievement (30). The down- side is that the prevalence of catheterization and stomal problems increases with the length of follow-up(24). Our study enrolled patients with long urethral strictures who had failed attempts of urethroplasty. Thus, the only alternative method that would make them catheter-free and continent was appendix diversion using Mitrofanoff principle. Although these patients need to perform CIC to empty their bladder, it does not interfere with their daily activities. The patients enrolled in our study suffered from com- plicated urethral stricture and were dependent on su- prapubic catheter for emptying their bladder ever since. After performing appendicular urinary diversion, these patients became catheter-free and did not have any dif- ficulties with intermittent catheterization for over two years. Our study reports a continence rate of 100% with good satisfactory results, consistent with the reports of previous articles(29-32). In our study, none of the six pa- tients had stomal stenosis during the fol¬low-up period. This means that our results were more satisfying than other studies.(17,24,33). The reason for a lower rate of stomal stenosis in our study might be the preservation of the mesenteric base of the appendix through wide reflection of the cecum with minimal manipulation which helped attain vas- cularity to decrease inflammation and mucosal dys- function. Also, a minimum tension was placed on the appendix between the bladder and the skin due to the appropriate selection of the location of the ostoma and, if necessary, the bladder was sutured to the rectus sheath. Using a suitable catheter size for insertion and then catheterization, appendix end speculation at the stoma site, as well as careful training of the catheteriza- tion technique could be other reasons. Although the results of appendix diversion are desira- ble, sometimes the appendix is not usable because of insufficient length or quality, short mesentery, or histo- pathologic changes consistent with chronic inflamma- tion or fibrous lumen obstruction(34). Regarding these situations, techniques such as the Monti method, or us- ing a bladder or cecal flap to partially span the distance between the bladder and abdominal wall are good alter- native methods.(13,35) CONCLUSIONS Based on the results of our study, Mitrofanoff technique is a valuable procedure with low incontinence and com- plication rates and should be considered in cases with unreconstructable urethral damage who cannot be treat- ed with other routine methods to achieve urinary conti- nence and low-pressure reservoir. ACKNOWLEDGEMENTS The authors are thankful for the assistance of the urol- ogy ward staff due to their invaluable help throughout this study. 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