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Urological Science 27 (2016) 177e180

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Urological Science

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Case report

Urothelial carcinoma arising within bladder diverticulumdReport of acase and review of the literature

Hung-En Chen, Yi-Chia Lin, Yi-Hong Cheng*

Division of Urology, Department of Surgery, Shin Kong Wu Ho-Su Memorial Hospital, Taipei, Taiwan

a r t i c l e i n f o

Article history:Received 30 July 2014Received in revised form10 March 2015Accepted 17 March 2015Available online 24 April 2015

Keywords:bladderdiverticulumurothelial carcinoma

* Corresponding author. Number 95, Wenchang RTaiwan.

E-mail address: yihong5229@yahoo.com.tw (Y.-H.

http://dx.doi.org/10.1016/j.urols.2015.03.0031879-5226/Copyright © 2015, Taiwan Urological Asscreativecommons.org/licenses/by-nc-nd/4.0/).

a b s t r a c t

Bladder diverticulum is an outpouching of bladder mucosa through the musculature of the bladder wall.The incidence of bladder diverticulum in Taiwan is about 1.7% in children and 23.4% in adults. Intra-diverticular carcinoma of urinary bladder is uncommon. It ranges from 0.8% to 14.3%. Here we report acase of urothelial carcinoma within a bladder diverticulum. A 60-year-old male patient had history ofBPH under medical treatment and right ureteral stone treated with extracorporeal shock wave lithotripsy(ESWL). He presented with painless gross hematuria about 3 months after ESWL. Intravenous pyelog-raphy showed a filling defect within the bladder diverticulum. Histopathological diagnosis of low gradeurothelial carcinoma arising from the bladder diverticulum was made following cystoscopic biopsy.Laparoscopic partial cystectomy was performed with subsequent intravesical chemotherapy. Tumorrecurrence was found not from the previous diverticulum but from another area during regularcystoscopy at the 6-month postoperative follow up. He underwent transurethral resection of bladdertumor. Pathology revealed a noninvasive, high grade urothelial carcinoma. There was no further bladdertumor recurrence during the 1-year follow-up period. Bladder-sparing surgery with close cystoscopyfollow up for intradiverticular urothelial carcinoma can be applied as an alternative treatment modality.Copyright © 2015, Taiwan Urological Association. Published by Elsevier Taiwan LLC. This is an open access

article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

1. Introduction

Bladder diverticulum is an outpouching of the urothelial mucosalining from the muscular network of the bladder.1 Bladder diver-ticula are either congenital or acquired, and are caused by a defectof the bladder wall and increased intravesical pressure.2 The lack ofa muscle layer in bladder diverticulum results in a loss ofcontractility, which results in urine stasis in the diverticulum.Chronic irritation of intradiverticular urine stasis may be the causeof chronic inflammation or infection. The potential risk of malig-nant neoplastic change inside the bladder diverticulum increases.3

Primary neoplasms arising within a bladder diverticulum areuncommon. The incidence ranges from 0.8% to 14.3%.4,5 It drawsattention because of the difficulty in early diagnosis and a higherrisk of early invasion. Poor prognosis has already beendocumented.1,3,6,7

We present a case of urothelial carcinoma arising within abladder diverticulum. Laparoscopic surgery with a bladder

oad, Shrlin Chiu, Taipei 111,

Cheng).

ociation. Published by Elsevier Ta

preserving procedure provides an alternative treatment modality.The relevant literature is reviewed.

2. Case report

The 60-year-old male patient, a chronic smoker, had a history ofBPH under regular medical treatment. Right flank pain occurredabout 2 years previously. Right ureteral stone with obstructiveuropathy and left renal stone was diagnosed. The right ureteralstone was successfully treated by extracorporeal shock wave lith-otripsy (ESWL). However, he presented with painless gross hema-turia about 2 months after ESWL. He had atypical cells in the urinecytology. Intravenous pyelography (IVP) revealed a filling defectwithin the bladder diverticulum (Fig. 1). Subsequent cystoscopyshowed multiple papillary tumors within a single diverticulumlocated at left posterior wall of bladder (Fig. 2). The size of thediverticulum measured about 3 � 3 � 3 cm3. A biopsy from thepapillary tumor specimen revealed a low grade papillary urothelialcarcinoma. Subsequent computed tomography also demonstratedmultiple tumors within the bladder diverticulum (Fig. 3). Therewasno evidence of lymphadenopathy, or distant or bony metastasis. Nobody weight loss was noted in the past year.

iwan LLC. This is an open access article under the CC BY-NC-ND license (http://

Fig. 1. Intravenous pyelography shows a sacculation from the left side of the urinarybladder. There is a filling defect within the bladder diverticulum.

Fig. 3. Contrast-enhanced abdominal computed tomography demonstrates a bladderdiverticulum protruding from the left posterior lateral wall with a soft tissue nodule inthe sac.

H.-E. Chen et al. / Urological Science 27 (2016) 177e180178

During admission, hematological and biochemical studies werewithin normal limits. Prostate specific antigen (PSA) was 1.49 ng/mL. Transurethral resection of bladder tumor was not feasible dueto narrowing of the diverticular neck. Therefore, he underwent

Fig. 2. Cystoscopic finding reveals papillary tumors arising within th

laparoscopic partial cystectomy. Cystoscopy was performed at firstto stent the left ureter with a temporary straight ureteral catheter. Afour-port transperitoneal approach was employed. It is not easy toidentify the diverticulum, due to its smaller size, under laparoscopy.Under the guidance of cystoscopic light from bladder below, thediverticulum was finally identified. After the diverticulum wasidentified, the bladder wall (about 1 cm lateral to the diverticulum)was grasped. The water in the bladder was emptied through thecystoscope. A Foley catheter was re-inserted to maintain intra-vesical low pressure, and prevented urine spillage during the par-tial cystectomy procedure. Partial cystectomy was then carried outto resect the entire diverticulum with margin. The specimen wasput in an Endo Catch bag immediately after complete resection. Thedistance from the resection margin to the diverticular neck is about0.4 cm in the direction of the orifice and 1 cm elsewhere in the

e bladder diverticulum located at the left posterior lateral wall.

H.-E. Chen et al. / Urological Science 27 (2016) 177e180 179

margin. The bladder was repaired with 3-O V-Loc suture materialusing a two-layer method. Three papillary tumors were noted in-side the bladder diverticulum in the gross specimen. Final histo-pathology revealed a high grade noninvasive papillary urothelialcarcinoma. The underlying stroma was not invaded. The resectionmargin showed focal urothelial hyperplasia and dysplasia withoutmalignancy (Fig. 4).

In total, six courses of postoperative intravesical chemotherapywith 30 mg mitomycin C was administered. At the 6-month followup, cystoscopy revealed multiple papillary tumor recurrence in theright lateral, left posterior and left lateral wall but away from theprevious diverticular site. The patient underwent transurethralresection of the bladder tumor. Histopathological charactersremained superficial, high grade urothelial carcinoma. Submucosainvasionwas not seen. Another 6 weeks of mitomycin C intravesicalchemotherapy was given. No further tumor recurrence was notedin a recent 1 year follow up.

3. Discussion

The incidence of bladder diverticulum varies from 1.7% to 13%.4

However, the incidence is much higher in recent reports and specialsubgroups of patients. Prakash et al5 reported the presence ofbladder diverticulum is 23.4% in cadavers. The prevalence ofbladder diverticulum is more common in men as compared withwomen (31.6% vs. 9%).5 Shakeri reported that the rate of bladderdiverticulum in patient with BPH is 48%. Bladder diverticula areeither congenital or acquired, and are caused by a defect of thebladder wall and increased intravesical pressure.2 The most likelysite of diverticulum formation is the area adjacent to the ureteralorifice where the longitudinal muscle fibers are absent and out-pouching is facilitated.

Deficiency or lack of a muscle layer in bladder diverticulummade it impossible to empty urine completely from the divertic-ulum. Chronic irritation of urine stasis inside the diverticulumresulted in chronic infection and inflammation and then facilitatedthe development of malignant neoplasms. In patients with bladderdiverticulum, mucosal inflammation, ulceration, dysplasia, squa-mous metaplasia, and leuoplakia was reported in 84% of patientswho received elective diverticulectomy.1 If a neoplasm developed

Fig. 4. Histopathological features revealing a section of bladder diverticular papillarytumor. The tumor shows papillary structures lined by layers of atypical urothelial cellswith crowding nuclei. Red arrows indicate urothelial carcinoma. Blue arrows indicatenormal uroepithelium. The tumor did not invade the lamina propia. There is no musclelayer in the bladder diverticulum. (For interpretation of the references to colour in thisfigure legend, the reader is referred to the web version of this article.)

in patient with bladder diverticulum, most of the neoplasm wouldbe located within the diverticulum. Carcinomas arising within thediverticulum are not uncommon. The incidence ranges from 0.8% to14.3%.4,5 Urothelial carcinoma was the most common type of ma-lignancy (78%), followed by squamous cell carcinoma (17%), coex-istence of urothelial carcinoma and squamous cell carcinoma (2%),adenocarcinoma (2%), and other rare tumors.8

Most bladder diverticula are asymptomatic. Neoplasms arisingwithin a bladder diverticulum poses difficulty in early diagnosis.The cardinal clinical presentation is painless gross hematuria fordiverticular tumor. According to Melekos et al,6 87.5% of patientswith bladder diverticular neoplasm presented with hematuria. Thediagnostic modalities is similar to that of bladder cancer. Urinecytology, cystoscopy, and radiologic examinations such as IVP,computed tomography or magnetic resonance imaging are usefulin the diagnosis of bladder diverticular cancer.

The modalities of surgical treatment varies from conservativetransurethral resection to aggressive radical cystectomy. Tran-surethral resection is a standard for removal of bladder tumor.However, the anatomic structure of bladder diverticulum made itimpossible to complete resection of bladder diverticular tumor insome cases such as where there was narrowing of the diverticularneck. It also poses risks of tumor spread from the more easilyruptured diverticular wall due to the lack of amuscular layer. Partialcystectomy is a good alternative for the treatment of bladderdiverticular cancer. With advances in minimal invasive surgery,laparoscopic diverticulectomy or partial cystectomy was graduallyaccepted by urologists in this clinical scenario with promising re-sults. In cases of Wang et al9 and ours, tumor recurrences awayfrom the previous diverticular site were all noted after laparoscopicpartial cystectomy. Both underwent transurethral resection ofbladder tumor and intravesical chemotherapy. The result was goodand no more recurrence were found until now.

The poor prognosis of bladder diverticular cancer was reportedin earlier studies. Kelalis and McLean1 reported that the averageperiod of survival was only 11 months in 19 cases of bladderdiverticular cancer. Faysal and Freiha3 also reported a low disease-free survival rate of about 8% in 11 cases with bladder diverticularcancer. They suggested radical cystectomy as a gold standard forpatients with bladder diverticular cancer. However, recent studieshave demonstrated better outcomes for these patients. Golijaninet al10 reported significant differences in 5-year disease-free sur-vival among patients with superficial tumors (83 ± 9%), superfi-cially invasive tumors (67 ± 7%) and extradiverticular disease(45 ± 14%).10 All of their 39 patients underwent transurethralresection of bladder diverticular cancer initially. The most impor-tant factor of prognosis is clinical stage, regardless of the histo-logical grade. They also mentioned favorable outcomes in anotherseries with 71% disease-free survival at 5 years in Baniel's series and89% 4-year disease specific survival in Garzotto's series with rela-tive conservative approaches.

In conclusion, conservative transurethral resection seems to beadequate for patients with superficial noninvasive urothelial car-cinoma with bladder diverticulum. Laparoscopic partial cys-tectomy is a favorable alternative for these patients and those whopose superficial invasive bladder diverticular carcinoma providedtransurethral resection is not feasible or complete. Close post-operative follow up is very important because of the high recur-rence rate.

Conflicts of interest

The authors declare that they have no financial or non-financialconflicts of interest related to the subject matter or materialsdiscussed in the manuscript.

H.-E. Chen et al. / Urological Science 27 (2016) 177e180180

Sources of funding

No funding was received for the work described in this article.

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