PEDIATRIC UROLOGY Suprapubic Percutaneous Assisted Cystoscopic Excision of Posterior Urethral Fibroepithelial Polyps in Pediatric Patients Esra Ozcakir*1, Mete Kaya1 Purpose: The aim of this study was to evaluate pediatric posterior urethral fibroepithelial polyps, their diagnosis and endoscopic treatments with suprapubic assisted transurethral polyp excision which is described by us. Materials and Methods: We reviewed the charts of patients (n=6) who underwent suprapubic percutaneous as- sisted cystoscopic excision for posterior urethral fibroepithelial polyp from 2014 to 2019. Their data were retro- spectively reviewed in terms of clinical features, diagnostic methods, endoscopic approaches, and postoperative results. Results: The 6 patients, the mean age of 3 years (4 months-6 years), with a solitary polyp of posterior urethra di- agnosed and removed by suprapubic percutaneous assisted cystoscopic excision in five years. The most common complaint was urinary tract infection (n:3). The urethral polyps were diagnosed by ultrasound and cystoscopy. There was no intraoperative or postoperative complication except for one patient with bleeding from the trocar site. All of the specimens after histopathology examinations showed fibroepithelial polyps and no recurrence was seen. Conclusion: Posterior urethral polyps may cause obstructive effect in the urinary tract. The treatment should be performed with the least possible invasive method without injuring urethral wall. We believe that suprapubic percutaneous assisted cystoscopic resection, described by us is an easy, reliable and effective method for treatment procedure of posterior fibroepithelial urethral polyps. Keywords: solitary urethral polyp; children; endoscopic resection; cystoscopy; voiding cystourethrography INTRODUCTION Isolated polyps can emerge and develop in any area of the urinary tract. The sources of their origination are mostly the ureter and renal pelvis, as well as the posterior urethra in rare cases(1). The posterior urethral fibroepithelial polyp (PUFP) is known as a rare and usually benign lesion emerging from the verumonta- num or posterior urethral mesodermal tissue(1-4). Polyps usually provoke a variety of explicit symptoms such as recurrent urinary tract infection (UTI), urinary reten- tion, and lower urinary tract symptoms among pediatric patients(1). To diagnose PUFP, ultrasound (US), voiding cystourethrogram (VCUG), and cystourethroscopy are used(3). The treatment approaches of PUFP include open cystostomy and transurethral resection or fulguration of the polyp by cystoscopy(3). The current study involves the six pediatric patients who underwent PUFP excision with suprapubic percutaneous assisted transurethral ex- cision (SPATE) endoscopically. In the present study, we aim to reflect on the experience of clinical appear- ance for PUFPs and treatment method by SPATE. MATERIALS AND METHODS This retrospective study is based on the evaluation of the data of patients diagnosed with PUFP who were treated in the pediatric surgery department of Universi- ty of Health Sciences, Bursa Medical Faculty between January 2014 and June 2019. The required data for the 1University of Health Sciences, Bursa Medical Faculty, Department of Pediatric Surgery, Bursa 16100, Turkey. *Correspondence: University of Health Sciences, Bursa Medical Faculty, Department of Pediatric Surgery, Bursa 16100, Turkey. phone: +90(224) 2955000. E-mail: dresramermer@hotmail.com. Received October 2019 & Accepted June 2020 analyses were obtained from the electronic database of the institution. The clinical characteristics, diagnostic methods, endoscopic approaches, surgical techniques, as well as postoperative results, were recorded for the cases with urethral polyp (Table 1). The data revealed that the endoscopically polyp excision by SPATE meth- od was applied to all patients by the same surgeon over the previous five years. Written informed consent was obtained from each parent before the procedure. This method is visually illustrated in Figure 1. Since the cys- toscopic approach is now a routinely practiced surgery for evaluation of various urinary diseases, we did not seek approval from the ethics committee because our method did not pose an additional risk for patients and is a modification of previous methods. All tissue sam- ples were examined histopathologically. Operative procedure SPATE: A smooth, mobile, pedunculated polyp devel- oping within posterior urethra was identified via cys- toscopy (Figure 2). During a cystoscopic operation, a 2 mm laparoscopic grasper was placed into the patient's filled bladder transvesically through a sharped-edged suprapubic bladder catheter's trocar. Mannitol solution was used for intermittent or continuous irrigation to get a brighter cystoscopic view. A laparoscopic instrument was utilized to hold polyp as tightly as possible. Stabi- lization of polyp secured total resection with ensuring a safe distance to the base of the bladder mucosa. In Urology Journal/Vol 18 No. 1/ January-February 2021/ pp. 86-91. [DOI: 10.22037/uj.v16i7.5688] Vol 18 No 1 January-February 2021 87 the meantime, a transurethral excision was performed easily by using the electrocautery resectoscope for the lesion's complete removal (Figure 3). The removed polyp was retrieved by transurethral basket catheter. Samples taken during the operation were examined his- topathologically. RESULTS The pediatric population included five boys and one girl aged between four months and six years (the mean age of 3). Of these, three patients (50%) had recurrent UTI with fever, vomiting, hematuria, and abdominal pain. Two patients had voiding issue with acute urinary re- tention and intermittent micturition resulting from pol- yp obstacle. The last boy presented with extra-urinary symptoms and had an incidentally detected urethral pol- yp by US. In addition, patients had concomitant urinary diseases such as vesicoureteral reflux, urinary collect- ing system duplication, and hydronephrosis. The pa- tients' characteristics and the diseases accompanied by PUFP were presented in Table 1. Diagnostic modalities for the PUFP were as followed: in two cases the poly- poid mass image was clearly identified by US (Figure 4), in three cases the polyp was detected by cystoscopy during vesico ureteral reflux (VUR) treatment, and for the last one, the VCUG revealed a polyp as a filling defect in the posterior urethra (Figure 5). All polyps, solitary and sizes were ranging from 5×8 mm to 9×20 mm emerged in the posterior urethra, and all of the pol- yps were removed successfully by SPATE. The urethral catheters were removed within 36 hours, following the surgical intervention. The extra surgical intervention included sub ureteric injection for three patients with VUR and ureteroneocystostomy in one patient who also has right-sided duplicated collecting system with VUR. Further, the clinical course of the patient with bi- lateral hydronephrosis was improved remarkably after SPATE procedure without extra intervention. Except for bleeding that stops spontaneously in the trocar line, no intraoperative or postoperative complications were observed. The mean duration of hospitalization was 3,4 days. Histopathologic examinations revealed fi- broepithelial polyps in all patients (Figures 6-7). The patients were continued to be monitored for a period of one year. They were followed up with the urinary US and urine analyses every month. Afterward, all pa- tients were evaluated by VCUG at the first 6th months and control cystoscopies at the first year. There were good cosmetic outcomes for all and no complications or recurrence was observed throughout this follow up period. DISCUSSION In childhood, PUFPs are rare lesions originating in the lower urinary tract, yet they are considered to be a benign fibrous tumour(3,4). Regarding the current liter- ature, the reported epidemiologic age of patients diag- nosed with PUFP ranges from first months to 80 years, whereas 82% of all cases usually occur in patients younger than 20 years and manifest themselves in the first decade(5-7). As claimed by Kimchhe and Downs, the average age of manifestation is between 8.25 and 9.7 years, yet our series of clinical studies represented the mean age of 3 years(6,7). Additionally, boys are known to have a higher risk of developing PUFP than girls; for all that, the literature and clinical data related to female patients are quite limited(3,8-10). Alike, in our series, male patients were the majority. The etiology of PUFP still remains unclear, and it is suggested that the pathology has congenital origins. In the literature, experts on PUFP propose certain con- cepts on the etiology of PUFPS, including the develop- mental failure in the invagination process of submucous glandular material of the prostate gland's inner zone, an abnormal protrusion of the urethral wall or epithelial changes secondary to the maternal estrogen(7,11,12). Be- sides, infectious, obstructive and traumatic causes have been theoretically considered as the physiological trig- gers of this disease(7,13). From the clinical viewpoint, PUFPs are accompanied with explicit symptoms due to intermittent or acute ob- struction of the bladder outlet, such as hesitancy, dimin- Percutaneous assisted cystoscopic excision of posterior urethral polyps -Ozcakir et al. Table 1: Patient’s demographic data (AD: Accompanied disease, AUR: Acute urinary retention, BH: Bilateral hydronephrosis, UNC: Ureteroneocystostomy, UTI: Urinary tract infection, R: Right side, SPATE: Suprapubic percutaneous assisted transurethral excision, VCUG:Voiding cystouretrography, VUR: Vesicoureteral reflux). Patient No Age Gender Symptoms Diagnose Size AD Treatment 1 4 month M UTI Cystoscopy 10x10mm R Grade III VUR SPATE 2 6 years M - US 20x9 mm - SPATE 3 4 years M Intermittent US 10x10 mm R Grade III VUR SPATE micturition AUR 4 5 years F UTI Cystoscopy 15x10 mm R Grade III VUR SPATE R Duplex System UNC 5 10 month M UTI VCUG 15x10 mm BH SPATE 6 2 years M Intermittent Cystoscopy 12x10 mm Bilateral Grade III VUR SPATE Figure 1. SPATE is illustrated ished urinary stream, incomplete emptying, and urinary retention. Hematuria and dysuria are other common symptoms associated with urinary tract infections(1,8). The clinical triad, including intermittent urinary re- tention, hematuria, and lower urinary tract symptoms, indicates highly probable and noticeable signs of ure- thral polyps in children(1,8). By reviewing the outcomes of 48 cases, Kearney et al. found obstruction (48%) to be the most common presenting symptom followed by hematuria (27%) and retention (25%)(14). Howev- er, PUFPs could also be asymptomatic depending on sizes and location of polyp. De Castro et al. reported on a series of 17 cases where patients had been treated throughout a 16-year period. In fact, 14 out of 17 cases were symptomatic, and three were identified and diag- nosed incidentally(8). In our series, the mostly observed presentation was reccurrent UTI (n:3), urinary retention with intermittent voiding (n:2), and the last one was asymptomatic. The diagnosis of PUFP is typically based on clinical findings, imaging modalities (US, VCUG) and cys- toscopic evaluation(3,4,7). While US tends to show polyps as an echogenic foci projecting into the lumen, VCUG reveals them as a polypoid filling defect, prolapsing through an external sphincter into the bulbous urethra. Intravenous urography is rarely applied because of ra- diation levels. Although US and VCUG are efficient diagnostic imaging, cystourethroscopy is utilised as a confirmatory strategy(3). Cystoscopic evaluation is the most specific examination for identifying polyps. Addi- tionally, because of the overlap in clinical presentation and radiological findings, diagnosis of polyp becomes a diagnostic challenge to distinguish a fibroepithelial pol- yp from a blood clot, radiolucent calculi or neoplasm. Cystoscopic evaluation; reveals the existence of polyps precisely and contributes to the excision of them simul- taneously. In addition, to overcome the overlap, CT and urine cytology could be helpful for the diagnosis of cal- culi and malignancy(11,13). Cystoscopy remains to be the most effective examination method, while the macro- scopic appearance of the polyp being diagnosed in the majority of cases represents solid, mobile, smooth and pedunculated fibrous tumours that originate from the vicinity of the verumontanum and lie along the surface Figure 2. Intraoperative cystoscopic view Figure 3. The cystoscopic view presented that the using of electrocautery resectoscope for the complete removal of the lesion. Percutaneous assisted cystoscopic excision of posterior urethral polyps -Ozcakir et al. Pediatric Urology 88 Vol 18 No 1 January-February 2021 89 of the posterior urethra(1,3). In our series, all polyps were observed on the posterior urethral wall by cystoscopy and confirmed by the histopathological examination. With reference to the current literature, a polyp can be accessed and removed by transurethral excision or open cystostomy(3,15-17). In 1985, Bruijnes et al. emphasized the importance of the general suprapubic approach through open cystostomy for the purpose of resecting polyps; in the meantime, it was mentioned that smaller lesions could be resected transurethrally(17). In addition, Schafer reported on three cases with intravesical ob- struction in 1989: two polyps were excised by open cys- totomy, and the last one was treated transurethrally(18). De Castro managed 17 cases of PUFP’s endoscopically without complications or relapses(8). Nowadays, open cystostomy is rarely required and can be applied mostly for removing large lesions. Common urethral polyps are usually removed transurethrally by the endoscopic resection electrocautery, cold knife or laser(1,2,6,7,11). In the present series, patients were successfully treated by SPATE procedure with the electrocautery for PUFP’s endoscopic resection. In addition, we relied on the transvesically suprapubic approach without conducting open surgery. Figure 4. US examination demonstrates that an irregular mass, arises from the bladder neck and extended into the bladder. Figure 5. VCUG image demonstrate that polypoid filling defect on bladder neck. Figure 6. Histopathologic examination shows polypoid lesion with papillary fronds lined by transitional urothelium and associat- ed submucosal fibrous stroma exhibiting mild chronic inflamma- tion (Hematoxylin and Eosin x 40). Figure 7. Histopathologic appearance with large magnification of polyp presents enriched stroma with various component, such as vascular formations with angiomatous features, smooth muscular fibers, pseudoglandular structures and inflammatory infiltrates (Hematoxylin and Eosin x 200). Percutaneous assisted cystoscopic excision of posterior urethral polyps -Ozcakir et al. It is important to note that to detect the original base of the polyp for the successful endoscopic treatment is sig- nificant. Although it is stated that the urethral approach can be easily performed in adults, it may not be able to reach the root of polyp cause of large peduncle that leads to the obstacle in childhood. Reaching the root of polyp might be complicated in some cases due to the obstacles that arise from the folding of the lesion's pe- dunculated body. Moreover, during cystoscopy, hyper- mobile pedunculated polyp might glide in and out of the bladder neck due to the endoscopic fluid flow. The use of a percutaneous dissector for the proper holding sim- plifies the identification of the polyp's root and helps to differentiate the urethral wall from the polyp itself dur- ing the resectoscopic excision. Thus, a full excision is secured under a direct vision without residual tissue. To the best of our knowledge, this is the first clinical series of children undergoing successful suprapubic assisted, transurethral polyp excision in terms of percutaneous treatment of PUFPs. A diagnosis of urethral polyps must be distinguished from inflammatory and neoplastic lesions located with- in the urethra. This usually includes non-opaque stones, a foreign body, posterior urethral valves, Cowper's duct cyst, ectopic urethral insertion, urethral diverticulum and/or hypertrophy of the verumontanum(14). These le- sions provoke outlet obstructions of different degrees, and thus they require the differential diagnosis to iden- tify a posterior urethral polyp(19). It is crucial to keep in mind that without accurate tissue diagnosis, the inva- sive open surgery management may cause a negative impact on the clinical follow-up, such as subsequent fibrosis or urethral strictures, thus requiring and leading to a second surgical operation(14). As the cystoscopy be- comes the gold standard for diagnosis, treatment has to be performed with the least possible invasive method to avoid precedents for injuring the urethral wall. In terms of SPATE, the proper holding of polyp guarantees a stretched appearance on the pedicle that allows the re- sectoscope to commit a complete excision without dam- aging the surrounding tissue. The SPATE procedure en- sures reliable outcomes from conducting an operation with direct observation of the process. In this sense, it is easier to perform as compared to other invasive strat- egies. In addition, this methodology requires no new or extra tools for SPATE, since all shaped-edged suprapu- bic bladder catheter's trocars are suitable and sufficient for use. In the case of children, polyps emerging from the ver- umontanum are congenital in origins(1,3,5,7,11,12). The ver- umontanum is viewed as a landmark for the exit of the mesonephric duct. Kuppusami and Moors conducted a histological analysis of the verumontanum to find that it incorporates smooth muscle and small glands; in all cases, it was lined by transitional cell epithelium(12). His- topathologically, PUFPs are characterised by a fibrous core covered by transitional epithelium. The edematous stroma becomes the most variable element which is often filled with various components, such as vascular formations with angiomatous properties, smooth mus- cular fibres, nerves, glands, pseudoglandular structures, and inflammatory infiltrates(5,8,12,13). Also, in our experi- ence, the histopathological examination confirmed the clinical diagnosis of fibroepithelial urethral polyps in all cases. There have not been reports on the malignant transfor- mation of PUFP to date. Nevertheless, the possibility of recurrence because of the incompletely removed polypoid lesion has been mentioned(20). Thus, we rec- ommend that close follow-up, even if the risk of recur- rence is low. Several urethral polyps have been associated with other congenital urinary tract anomalies(7,14,19). For example, De Castro reported that 50% of the patients with ure- thral polyps had a urinary pathology of different nature, especially VUR(8). Kimche and Lask reported about 50 cases with a similar clinical picture; dilatation of the upper collecting system was identified in 10 patients (20%), while VUR was diagnosed in four (8%) pa- tients(6). Additionally, bladder diverticulum was present in four patients (8%). In the series of 18 patients report- ed by Akbarzadeh et al., six patients had VUR (33%), and five patients had dysfunctional voiding findings (27%)(19). In our series, the associated diseases involved duplicated urinary system in one patient and VUR in four patients (66,6%). Drawbacks in our study concerning SPATE include the limited number of cases, a low degree of control over cystoscopic evaluation and the shortage of long-term uroflowmetry results after the procedure. However, our initial observations presume that the SPATE procedure is easy to perform, which makes it also efficient for complete excision without residue. CONCLUSIONS PUFP is a rare pathology in children, whereas cystosco- py remains the gold standard for diagnosing the disease. Despite the status of a benign lesion, urethral polyps may cause obstructive effect in the urinary tract. PUFP resection by endoscopic methods should be considered a complete treatment in the majority of patients. The current research review has revealed that tran- surethral resection is the best treatment choice, while SPATE is the most reliable strategy for ensuring com- plete excision. ACKNOWLEDGEMENTS We would like to thank Duncan T. Wilcox, the chief of Pediatric Urology at Colorado University, Children's Hospital Colorado, for kindly reviewing our manu- script. CONFLICT OF INTEREST The authors declare that they have no competing inter- ests. REFERENCES 1. Gleason PE, Kramer SA. 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