Understanding an unusual urothelial disorder: cystitis cystica et glandularis

Objectives: To study the presentation and natural course of cystitis cystica et glandularis. Methods: A retrospective analysis of patients with histopathologically confirmed cystitis cystica et glandularis from March 2016 to March 2018 who at least completed their 2 years’ follow-up was performed. Perioperative details along with the last available follow-up were included in the analysis. Results: A total of 10 patients were included. The mean age (± standard deviation) was 33.4 (±14.0) years and nine (90%) were men. The most common presentation was storage and voiding lower urinary tract symptoms (80%) along with haematuria (40%) and dysuria (20%). Four patients had the presence of hydronephrosis in preoperative imaging, of which three patients had bilateral mild hydroureteronephrosis. All the patients underwent transurethral resection of the bladder tumour as all were diagnosed with urinary bladder mass on preoperative imaging. All the patients had a trigonal lesion with a bullous appearance partially obstructing the bladder neck. Six patients underwent double J stenting in the perioperative period. The mean (± standard deviation) follow-up duration was 32.8 (±7.5) months. Patients were kept on regular surveillance with imaging and cystoscopy as indicated. Eight patients (80%) developed recurrence in the follow-up period. The mean number of recurrences was 1.5 (±1.1). One of the patients had to undergo augmentation ileocystoplasty with bilateral ureteric reimplantation because of the recurrent lesion with small contracted bladder, while another patient underwent cystectomy with urinary diversion owing to recurrence and refractory lower urinary tract symptoms. Besides, there was no evidence of malignancy after this entity in any of the patients. Conclusion: Cystitis cystica et glandularis is a rare clinic pathological entity which often mimics bladder tumour. Cystitis cystica et glandularis is common in men and often presents with lower urinary tract symptoms. Transurethral resection forms the mainstay of treatment. However, it is often associated with upper tract hydronephrosis. Its controversial premalignant nature compounded with recurrence and risk of upper tract deterioration warrants close surveillance. Level of evidence: 4

Primary vesical actinomycosis with changes of cystitis cystica presenting as bladder mass

Actinomycosis is a rare chronic granulomatous suppurative infection caused by Gram-positive bacteria. The occurrence of primary vesical actinomycosis is extremely rare and only a few cases have been reported. Pre-operative diagnosis of vesical actinomycosis is challenging as the clinical and radiological features usually point towards bladder malignancy. Therefore, in most cases, definitive diagnosis is usually made after histopathological examination of the involved tissue. A 60-year-old male presented with complaints of hematuria, burning micturition, irritative, and obstructive urinary symptoms for 15 days. USG revealed a large soft-tissue mass having a polypoidal intraluminal and extraluminal component and involving the right posterolateral urinary bladder wall. CT scan showed a large irregular soft-tissue mass with multiple cystic lesions involving the right lateral wall of the urinary bladder. Transurethral resection of bladder mass biopsy was performed and the histopathological examination showed bacterial colonies of Actinomyces with changes of cystitis cystica. The patient was treated with amoxicillin and potassium clavulanate for 3 months. Actinomycosis should be kept as a rare differential diagnosis in cases presenting as bladder mass. The diagnosis is most commonly made by histopathology and may need a repeat biopsy to arrive at the correct diagnosis. The patient should be treated by penicillin group of antibiotics for 2–3 months and followed up for years to detect any recurrence.

Florid cystitis cystica et glandularis causing irreversible renal injury

Cystitis cystica et glandularis (CCEG) is widely believed to be innocuous and self-limiting. We report a case of a 32-year-old male patient who was found to have gross bilateral hydroureter and hydronephrosis and an estimated glomerular filtration rate of 3 ml/min/1.73 m2. Cystoscopy revealed extensive cystic and nodular lesions involving most of the bladder urothelium, which proved to be CCEG on histopathological analysis. Retrograde and anterograde stents could not be inserted due to obstruction of the ureters at the level of the vesicoureteric junction. Percutaneous nephrostomies were subsequently inserted. Although there was evidence of improvement of the CCEG on follow-up cystoscopy, no improvement of renal function, despite decompression with percutaneous nephrostomies, was seen. He was subsequently placed on the waiting list for a renal transplant. We believe this to be the only known case reported of florid CCEG obstructing the upper urinary tracts bilaterally, causing irreversible renal injury.

Vesicular Cystitis: A Chronic Cystitis Variant Often Unresponsive to Antibiotics and Electrofulguration

<b><i>Objective:</i></b> The aim of the study was to report on the presentation and outcomes of vesicular cystitis (VC), a chronic cystitis exhibiting translucent bladder mucosal vesicles, among women with antibiotic-refractory recurrent urinary tract infections (RUTIs). <b><i>Methods:</i></b> An analysis of our Institutional Review Board-approved series on antibiotic-refractory RUTIs was performed, selecting for documented VC lesions on cystoscopy. All patients had RUTIs defined as ≥3 urinary tract infections/year with positive urine culture. All patients were extensively treated with antibiotics with no resolution of RUTIs and were offered electrofulguration (EF) of VC lesions under anesthesia as a last resort. All patients had a 6-month post-EF office cystoscopy documenting persistence or resolution of the lesions, and a clinical outcome assessment based on RUTI frequency. <b><i>Results:</i></b> Of 482 patients, 18 (3.7%) treated during 2011–2017 met the study criteria. VC was most commonly found over the dome/anterior wall (7/18, 38%) and as pancystitis (7/18, 38%). There was often concomitant cystitis cystica of the trigone (8/18, 44%). At post-EF cystoscopy, persistence of VC was noted in 10/18 (56%) patients; 6/18 (33%) underwent repeat EF and an additional 3/18 (17%) were retreated due to new lesions after initial resolution. Two (11%) patients required simple cystectomy and urinary diversion due to RUTIs refractory to all interventions. Within a median follow-up of 2.8 years after EF, clinical cure was observed in 5/18 (28%), improvement in 10/18 (56%), and failure in 3/18 (17%) patients. <b><i>Conclusions:</i></b> Among women with antibiotic-refractory RUTIs, VC is an infrequent and persistent form of cystitis with a predilection for non-trigonal bladder surfaces, whose management is challenging.