Cystitis glandularis with perivesical lipomatosis

CYSTITIS GLANDULARIS PERIVESICAL

WITH

LIPOMATOSIS

Frequent Association of Two Unusual Proliferative Conditions

SUBBARAO MILTON

V. YALLA, M.D.

IVKER,

M.D.

HARRY M. BURROS, FRED

DOREY,

M.D.

M.D.

From the Department of Urology, Graduate Hospital of the University of Pennsylvania, Philadelphia, Pennsylvania, and Underwood Memorial Hospital, Woodbury, New Jersey

ABSTRACT - Review of literature on patients with pelvic lipomatosis showed an increased incidence of proliferative cystitis, notably cystitis glandularis. With the report of 2 of our cases, the incidence appears to be as high as 75 to 80 per cent. The pathologic interrelationship of these two proliferative conditions is discussed.

Cystitis glandularis, a rare and severe form of proliferative cystitis, is known to occur secondary to changes in extrophied bladder, chronic irritation due to infection or obstruction, and persistence of urachal elements. 1,2This unusual form of cystitis has also been found in association with perivesical lipomatosis which is also known as pelvic lipomatosis. The latter condition is an unusual perivesical proliferative process involving the mature fatty tissue of the pelvic retroperitoneal space. 3-11The pelvic viscera are involved in varying degrees. More than 35 cases were reported and many more although recognized were probably not included in the literature. Only 8 patients among these cases had successful cystoscopic evaluation and bladder mucosal biopsy. The remaining patients did not have endoscopic studies because of technical difficulties secondary to extensive deformities of the posterior urethra and base of the bladder. Six of the 8 patients who had endoscopic evaluation were found to have cystitis glandularis and/or other variants of proliferative cystitis. Cystitis cystica and cystitis follicularis are the other two forms.

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We submit case reports of 2 patients who had these proliferative conditions in association with each other. Our purpose is to bring attention to the clinical association of these two relatively rare conditions. A review of the literature is made, and a discussion on pathologic interrelationship is presented. Case Reports Case 1 A forty-year-old black man was first seen in September, 1971, with a history of pain in the lower abdomen associated with nausea and vomiting. Urinalysis showed bacteria and pus cells. He was known to have had mild systolic hypertension which was controlled with tranquilizers. Initial blood pressure was 170/90 mm. Hg, and in a few weeks it stabilized at 140/90 mm. Hg. Blood chemistry was normal and the urine culture negative. Findings on physical examination were essentially normal. Rectal examination revealed firm and irregular prostate, the upper limits of which could not be properly delineated. The prostate appeared to have been displaced

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FIGURE 1. Case 1. (A) Scout film showing radiolucent perivesical lipomatous tissue. (B) Excretory urogram revealing bilateral ureterectasis with severe degree of obstruction on left side; upward displacement of base of bladder with vertical elongation are also demonstrated.

superiorly. Scout film showed a soft tissue shadow of the bladder around which there was a radiolucent halo of 2 to 2.5 cm. in width (Fig. 1A). Excretory urogram demonstrated bilateral ureteral obstruction, vertical elongation of the bladder, and upward displacement of the base (Fig. 1B). Cystoscopic findings were elongation and anterior displacement of the posterior urethra and edematous and polypoidal bladder mucosa. The ureteric orifices could not be identified. Histologic findings of the polypoidal lesions were consistent with cystitis glandularis. Epithelial hyperplasia and attempts of gland formation were noteworthy features. Urinary cultures for tuberculosis were repeatedly negative. A left ureteroneocystostomy was performed since the patient continued to experience left flank discomfort. Follow-up evaluation with cystoscopy and bladder mucosal biopsy showed cysti tis follicularis, cystitis cys tica, and cystitis glandularis. This patient never experienced significant voidingproblems although severe deformity and elongation of the posterior urethra were noted. He was warned of the progressive involvement of the upper urinary tract and possible supravesical diversion.

normal as were all laboratory work-ups. Intravenous urogram revealed evidence of obstruction at the ureterovesical junction. Biopsy of the proliferative lesions of the bladder demonstrated cystitis glandularis with enteric metaplasia. Barium enema was normal. Cystoscopy was performed in April, 1971, and the bladder lesions were resected again. Although he was voiding better, his blood urea nitrogen was elevated to 25 mg. in November, 1971. Increased obstruction to the left ureter and dilation of the distal right ureter were noted with excretory urogram (Fig. 2). Urinalysis and urine cultures were repeatedly normal. In March, 1972, bilateral ureteroneocystostomy was performed since he showed progressive hydroureteronephrosis and experienced right flank pain. Exploration also revealed pelvic lipomatosis. Barium enema in September, 1973, revealed posterior deviation of proximal rectum and distal sigmoid. Intravenous urogram showed progressive increase in hyroureteronephrosis. This patient was informed that eventually he would require supravesical diversion.

Case 2

Clinically, cystitis glandularis is often mistaken for a malignant process, and it is usually diagnosed with cystoscopic and histologic evaluations. Histologic appearance reveals glandular replacement of the submucosal region with or without inflammatory reaction. ’ Patch12 believed that leukoplakia and glandular changes were the result of epithelial metaplasia in response to a call for

A forty-one-year-old white man was seen in February, 1971, with a two-year history ofpainful ejaculations, narrow urinary stream, and nocturia. Findings on physical examination were essentially normal. Rectal examination, however, showed thickened and fixed areas in the vicinity of the seminal vesicles. Prostatic biopsies were

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Comment

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altered function or at least as a result of altered environment. It was suggested that the extent and intensity of metaplastic process were in direct relation to the intensity and duration of the exciting factor. Kittredge and Brannan13 found that most of their reported cases, in contrast to others, were not associated with pyuria and symptoms of vesical irritability. They doubted if chronic irritation of vesical mucosa was actually necessary for glandular metaplasia. The fact that chronic inflammatory reaction was present without evidence of urinary tract infection was considered to represent a local response of tissue to an altered environment such as one would encounter in association with neoplastic processes. The increased rate of occurrence of cystitis glandularis in patients with pelvic lipomatosis suggests that the latter could be responsible in initiating the glandular metaplasia. It is also possible that some other etiologic agents could initiate these two proliferative conditions simultaneously. The cause of pelvic lipomatosis remains uncertain although several different opinions were expressed. Rosenberg et nl. E described this process as a manifestation of Dercum’s disease (adiposis it to be a localized dolorosa). 0th ers considered form of obesity. Microscopic examination of the lipomatous tissue showed mature fatty cells with or without inflammation. Pelvic lipomatosis is seen in many forms. Barry, Bilbao, and Hodges9 made a detailed review of the literature and found that only one half of the patients with pelvic lipomatosis had any significant difficulties with voiding despite the gross deformities of the posterior urethra and base of the bladder. Cystoscopy was not possible in the majority of these patients because of unusual elongation of the posterior urethra. The radiologic manifestations are more characteristic. The classic signs are: (1) radiolucent areas of lipomatous tissue in the bony pelvis, (2) elevation and vertical elongation of the bladder with an inverted teardrop or gourd appearance, and (3) straightening of the rectosigmoid with or without extrinsic compression. These 2 patients had a severe degree of bilateral ureteral obstruction and extensive cystitis glandularis in the vicinity of the ureteric orifices. Radiologic studies demonstrated obstruction at the ureterovesical junctions and medial deviation of the distal ureters. Pepper, Clemmett, and Drew7 similarly observed that the ureteral obstruction was caused by cystitis glandularis. Our 2 patients were able to empty their bladders

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Case 2. Excretory urogram exhibiting FIGURE 2. marked degree of bilateral ureter-al obstruction at ureterovesical junction. Vertical elongation and upward displacement of base of bladder can be recognized.

although unusual elongation and compression on the posterior urethra were recognized cystoscopically. The second patient experienced painful ejaculation; this particular symptom was not previously reported in patients with pelvic lipomatosis. This patient, in addition, had a moderate degree of voiding difficulty. It is unlikely that obstruction at the vesical outlet alone could have caused cystitis glandularis since it is not the usual experience of the urologist to encounter glandular metaplasia in patients with prostatism or urethral strictures. Cystitis glandularis in all these patients with pelvic lipomatosis could not have been caused entirely by infection or obstruction to the POSterior urethra. The perivesical lipomatous compression in the region of the base of the bladder could have created mucosal and submucosal edema which in turn produced epithelial and glandular metaplasia. Radiologically greater vesical deformities occurred at the base and inferior segments of the bladder where the proliferative mucosal changes appeared with great frequency. Submucosal edema as the result of lymph and

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venous stasis in this region could create an altered environment rich in protein fluid. Chronic venous obstruction in the pelvis with marked collateral formation has been recently described in a patient with pelvic lipomatosis. l4 Similar changes in the venous system probably existed in other cases of pelvic lipomatosis. The edematous fluid could be an excellent nutritive medium for tissue proliferation.‘5s16 Elephantiasis characterized by epiderma1 changes and connective tissue hyperplasia is a classic example of changes secondary to venous and lymphatic obstruction. Unlike its base and lower segment, the dome of the bladder, although encased by the lipomatous tissue, still retains its distensibility. Cystograms, therefore, demonstrate pear- or gourd-shaped deformities. The limits of distensibility in the region of the base of the bladder are probably restricted by the compromised perivesical space with lipomatous tissue. The large amounts of fatty tissue between the lower segment of the bladder and the bony walls of the true pelvis and pelvic floor would cause constant compression on the walls of the bladder and might create venous and lymphatic obstruction. These impressions are consistent with the suggestions of Kittredge and Brannanr3 who believed that cystitis glandular-is could occur as a reaction to an altered environment. We also suggest a majority of the patients with pelvic lipomatosis who could not be subjected to cystoscopic evaluations for technical reasons were probably harboring changes of prohferative cystitis. Conversely, it is also possible that cystitis glandular-is may be associated with varying degrees of lymphatic or venous obstruction in the pelvis.

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Veterans Administration Hospital 1400Veterans of Foreign Wars Parkway

West Roxbury, Massachusetts 02132 (DR. YALLA) References Urological Pathology, Philadelphia, Lea & Febiger, 1952, vol. 1, p. 220. 2. STIRLING, C., and ASH, J. E.: Chronic proliferative lesions of the urinary tract, J. Ural. 45: 342 (1941). 3. ENGELS, E. P.: Sigmoid colon and urinary bladder in high fixation, Radiology 72: 419 (1959). 4. FOGG,L. B., and SMYTH,J. W.: Pelvic hpomatosis: a 1. HERBUT, P. A.:

condition simulating pelvic neoplasm, ibid. 90: 558 (1968). 5. MAHLIN, M. S., and DOVITZ, B. W.: Perivesical hpomatosis, J. Ural. 100: 720 (1968). 6. LUCEY, D. T., and SMITH, M. J. V.: Pelvic lipomatosis, ibid. 105:341 (1971). 7. PEPPER, H. W., CLEMMETT, A. R., and DREW, J. . Pelvic lipomatosis causing urinary obstruction, Br. F’kadiol. 44: 313 (1971). 8. ROSENBERG, B., HURWITZ, A., and HERMANN, H.: Dercum’s disease with unusual retroperitoneal and paravesical fatty infiltration, Surgery 54: 451(1963). 9. BARRY, J. M., BILBAO, M. K., and HODGES, C. V.: Pelvic lipomatosis: a rare cause of suprapubic mass, J. Ural. 109: 592 (1973). 10. MALTER, I. J., and OMELL, G. H.: Pelvic lipomatosis in a woman, Obstet. Gynecol. 37: 63 (1971). 11. CARPENTER,A. A.: Pelvic lipomatosis: successful surgical treatment, J. Urol. 110: 397 (1973). 12. PATCH, F.: Epithelial metaplasia of the urinary tract, J.A.M.A. 136: 824 (1948). 13. KITTREDGE, W. E., and BRANNAN,W.: Cystitis glandularis, J. Urol. 81: 419 (1959). 14. SCHECHTER, L. S.: Venous obstruction in pelvic lipomatosis, ibid. 3: 757 (1974). 15. BEESON, P. B., and MCDERMOTT, W., Eds.: CecilLoeb - Textbook of Medicine, 12th ed., Philadelphia, W. B. Saunders and Co., 1967, p. 743. 16. ROBBINS,S. L.: Pathology, 3rd ed., Philadelphia, W. B. Saunders and Co., 1967, p. 139.

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