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International Urology and Nephrology 33: 635–636, 2001. © 2002 Kluwer Academic Publishers. Printed in the Netherlands. 635 Cystitis cystica glandularis masquerading as a bladder tumor Iqbal Singh & M.S. Ansari Department of Urology, All India Institute of Medical Sciences, New Delhi-29, India Abstract. A young man admitted with complaints of painless paroxysmal gross hematuria was found to have bilateral vesical masses involving the bladder base, trigone and ureteric orfices. Urine cytology and AFB were negative. Transurethral resection of the bladder mass was carried out and the histopathology suggested cystitis cysica glandularis. The literature regarding this entity has been reviewed and the differential diagnosis discussed. Key words: Cystitis, Cystitis cystica, Cystitis glandularis, Tuberculous cystitis Case report A 30-year-old male presented with complaints of multiple episodes of gross painless hematuria for over 12 months. Routine blood parameters were within normal limits. Urine cytology was unremarkable and the urinary polymerase chain reaction (PCR-TB) and urine for AFB were negative. USG and color flow suggested a horseshoe kidney with a vascular kissing lesion along the postero-inferior wall with a growth in the urinary bladder involving both lateral walls suggestive of transitional cell carcinoma. CT evalu- ation confirmed an irregular bladder wall thickening and mass lesion in the same region (Figure 1). Cystoscopic evaluation revealed a diffuse papil- loma involving the bladder base trigone and both ureteric orifices, which could not be visualized. Transurethral resection of the bladder tumor was carried out and the histopathology was reported as non-specific chronic inflammation with necrotic granulomas, and no evidence of malignancy, sugges- tive of cystitis cystica and glandularis. Based on this report an empirical four drug anti tubercular therapy (ATT) was started. Following subjective improvement a full 12-month (4+8) course of ATT was continued. Six month post therapy IVU revealed polypoidal filling defects arising from both the lateral walls of the bladder with infiltration of the left ureterovesical junction by the bladder mass and a normally functioning HSK (Figure 2). A renal dynamic scan showed bilaterally normally functioning kidneys with no evidence of stasis. The patient is doing well and he continues to be on a 6 monthly DTPA-RDS follow up with no evidence of obstruc- tion. Discussion The term cystitis cystica is a misnomer and reflects the unfortunate habit of labelling obscure bladder condi- tions as cystitis cystica (CC) [1]. Though (CC) and cystitis cystica glandularis (CCG) are by no means uncommon but the fact that these conditions may mimic a bladder tumor has not been well described in literature. An accurate pre operative diagnosis is not always feasible since the clinical and radiological findings may frequently be deceptive as in the present case. However at times urine cytological washings may enable a correct preoperative diagnosis by an astute pathologist [2]. Cystitis cystica (CC) is an interesting urothelial pathology whose etiology, morphology and clinical significance are poorly understood [3]. It may involve the human urinary bladder and trigone in both sexes. It is known to occur in certain chronic inflammatory infections of the bladder in children and adoles- cents. The exact connotations of CC are confusing in the literature. According to some it is a benign bladder epithelial abnormality while for others CC is considered to be a premalignant disease of the urinary bladder. Very few reports document the progression from cystitis cystica and glandularis to carcinoma of the bladder [4]. According to Olsen et al. [4] CC

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International Urology and Nephrology 33: 635–636, 2001.© 2002 Kluwer Academic Publishers. Printed in the Netherlands.

635

Cystitis cystica glandularis masquerading as a bladder tumor

Iqbal Singh & M.S. AnsariDepartment of Urology, All India Institute of Medical Sciences, New Delhi-29, India

Abstract. A young man admitted with complaints of painless paroxysmal gross hematuria was found to havebilateral vesical masses involving the bladder base, trigone and ureteric orfices. Urine cytology and AFB werenegative. Transurethral resection of the bladder mass was carried out and the histopathology suggested cystitiscysica glandularis. The literature regarding this entity has been reviewed and the differential diagnosis discussed.

Key words: Cystitis, Cystitis cystica, Cystitis glandularis, Tuberculous cystitis

Case report

A 30-year-old male presented with complaints ofmultiple episodes of gross painless hematuria for over12 months. Routine blood parameters were withinnormal limits. Urine cytology was unremarkable andthe urinary polymerase chain reaction (PCR-TB) andurine for AFB were negative. USG and color flowsuggested a horseshoe kidney with a vascular kissinglesion along the postero-inferior wall with a growthin the urinary bladder involving both lateral wallssuggestive of transitional cell carcinoma. CT evalu-ation confirmed an irregular bladder wall thickeningand mass lesion in the same region (Figure 1).

Cystoscopic evaluation revealed a diffuse papil-loma involving the bladder base trigone and bothureteric orifices, which could not be visualized.Transurethral resection of the bladder tumor wascarried out and the histopathology was reportedas non-specific chronic inflammation with necroticgranulomas, and no evidence of malignancy, sugges-tive of cystitis cystica and glandularis.

Based on this report an empirical four drug antitubercular therapy (ATT) was started. Followingsubjective improvement a full 12-month (4+8) courseof ATT was continued. Six month post therapy IVUrevealed polypoidal filling defects arising from boththe lateral walls of the bladder with infiltration ofthe left ureterovesical junction by the bladder massand a normally functioning HSK (Figure 2). A renaldynamic scan showed bilaterally normally functioningkidneys with no evidence of stasis. The patient is

doing well and he continues to be on a 6 monthlyDTPA-RDS follow up with no evidence of obstruc-tion.

Discussion

The term cystitis cystica is a misnomer and reflects theunfortunate habit of labelling obscure bladder condi-tions as cystitis cystica (CC) [1]. Though (CC) andcystitis cystica glandularis (CCG) are by no meansuncommon but the fact that these conditions maymimic a bladder tumor has not been well describedin literature. An accurate pre operative diagnosis isnot always feasible since the clinical and radiologicalfindings may frequently be deceptive as in the presentcase. However at times urine cytological washingsmay enable a correct preoperative diagnosis by anastute pathologist [2].

Cystitis cystica (CC) is an interesting urothelialpathology whose etiology, morphology and clinicalsignificance are poorly understood [3]. It may involvethe human urinary bladder and trigone in both sexes.It is known to occur in certain chronic inflammatoryinfections of the bladder in children and adoles-cents. The exact connotations of CC are confusingin the literature. According to some it is a benignbladder epithelial abnormality while for others CC isconsidered to be a premalignant disease of the urinarybladder. Very few reports document the progressionfrom cystitis cystica and glandularis to carcinoma ofthe bladder [4]. According to Olsen et al. [4] CC

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Figure 1. Cystographic phase of an excretory pyelographic filmshowing infiltration of the left ureterovesical junction by the bladdermass causing kinking of the left ureter (→).

Figure 2. CT scan showing an irregular bladder wall thickening andmultiple polypoidal masses (→) arising from both lateral walls ofthe bladder in the base and trigone.

reflects mobilization of the humoral immune defensemechanism in response to various agents, among thesea sub clinical malignant tumor. Cystitis cystica glandu-laris on the other hand is a form of proliferativecystitis, which can be mistaken for a tumor clinically,and radiologically [2].

The present case was confused with a bladdertumor since the trigonal mass had infiltrated one of theostia leading to hydroureteronephrosis. Though this is

known [5] to occur the possibility of an underlyingbenign etiology always seems to be remote and wayback in the treating clinicians mind.

The possible cause of CCG in the present casecould have been an underlying tuberculous infec-tion either of the genitourinary tract or elsewhere inthe body, which could have lead to certain humoralchanges causing a proliferative cystitis. Thoughwe failed to establish a microbiological/serological/cytological evidence of tuberculous infection in thebody we persisted with empirical antituberculartherapy since he seemed to have completely improved.Other known causes of CC/CCG documented in liter-ature include bladder calculi [6], chronic catheterisa-tion in paraplegics, bladder schistosomiasis [7], andchronic bacterial cystitis [8] leading to metaplasticcystitis [9].

The present case serves to remind and alertthe urologist to certain benign chronic inflammatoryconditions like proliferative cystitis mimicking infil-trating bladder tumors.

References

1. Sarma KP. Cystitis cystica (cystosis) with bladder cancer. J Urol1978; 120(2): 169–171.

2. Kapila K, Verma K. Cytology of cystitis cystica glandularis ofthe bladder – a case report. Indian J Pathol Microbiol 1996;39(4): 303–305.

3. Jost SP, Dixon JS, Gosling JA. Ultrastructural observations oncystitis cystica in human bladder urothelium. Br J Urol 1993;71(1): 28–33.

4. Olsen B, Johansen TE, Majak BM. Cystitis cystica – a prema-lignant condition? Tidsskr Nor Laegeforen 1993; 10; 113(18):2255–2256.

5. Daszkiewicz E, Musierowicz A. Cystitis cystica in the form ofa tumor infiltrating the ureteral ostium. Pol Przegl Chir 1971;43(10): 1699–1702.

6. Fein RL, Winton L, Gomez RR, Needell MH. Bladder calculienveloped by extensive cystitis glandularis: case report. J Urol1983; 130(3): 558–559.

7. Zahran MM, Kamel M, Mooro H, Issa A. Bilharziasis of urinarybladder and ureter: comparative histopathologic study. Urology1976; 8(1): 73–79.

8. Belman AB. The clinical significant of cystitis cystica in girls:Results of a prospective study. J Urol 1978; 119(5): 661–663.

9. Erturk E, Erturk E, Sheinfeld J, Davis RS. Metaplastic cystitiscomplicated with Von Brunn nests, cystitis cystica, andintestinal type of glandular metaplasia. Urology 1988; 32(2):165–167.

Address for correspondence: Dr Iqbal Singh, M.S., D.N.B., SeniorResident (urology) AIIMS, F-14 South Extension Part-2, NDSE-2,New Delhi-110049, IndiaE-mail: [email protected]