Ureteritis Cystica Causing Obstruction

DEBA P. SARMA, MD, and JOHN H. LUNSETH, MD,t New Orleans, La has been known about at least since 1761, when Morgagni described it grossly in autopsy cases.1 During the past four decades, it has been recognized as a cause of ureteral obstruction and has been frequently clinically diagnosed by means of retrograde pyelography, wherein the cysts cause punched out areas or a ragged outline, depending on their size.2 Our case is unusual because, unlike most of the other reported cases, the ureteral obstruction was not caused by the cystic component, but by a localized area of submucosal fibrosis containing a cluster of Brunn's epithelial nests.
URETERITIS CYSTICA
CASE REPORT

A 58-year-old black man with a long history of peptic ulcer disease was admitted for evaluation of recent gastrointestinal hemorrhage. For the past five years, he had also had episodes of severe grabbing pain in the left costovertebral angle, lasting up to 30 minutes. He denied hematuria or ever passing a stone in the urine. He had taken reserpine for four years to control high blood pressure. On admission, the serum BUN level was 27 mg/dl and creatinine level. 2.6 mg/dl. Urine cultures were negative. An intravenous pyelogram and a retrograde pyelogram (Fig 1) showed dilatation of "the left renal pelvis and calyces with partial obstruction of the ureteropelvic junction. No filling defect was noted. During retrograde pyelography, the patient had severe pain in the left flank, simulating his past episodal pain. Angiograms failed to reveal any abnormal vessels. At surgical exploration, the left ureter was found to be kinked immediately below the ureteropelvic junction, and small, dense fibrous bands were dissected off the ureter at that point. A Whitaker test of urine flow showed positive readings at 12.0 and 4.4 ml/min. The ureteropelvic junction along with a short segment of the adjacent renal pelvis and the ureter were resected, and an end-to-end anastomosis was done. The postoperative course was essentially uneventful. During the follow-up period of four months, the patient had no renal pain, but retrograde pyelograms were similar to those obtained preoperatively. though there was faster flow of dye through the ureteropelvic junction. The resected ureter measured 1.5 cm in length and 0.4 cm in diameter, and the lumen was narrowed by a projecting mass of fibrous tissue containing Brunn's epithelial nests (Fig 2). The nests were composed of transitional or squamoid cells forming only a fewrudimentary cysts. The proliferating fibrous stroma was devoid of inflammatory cells, and the overlying ureteral transitional epithelium was normal.
DISCUSSION

obstruction caused by cysts projecting into the lumen and producing characteristic filling defects on retrograde pyelography.2 Although it is commonly agreed that ureteritis cystica is secondary to chronic irritation,1'3 we found no evidence of the usual irritants, namely, ureteral calculus and pyelonephritis. The lack of inflammatory exudate, however, does not conflict with the diagnosis of ureteritis cystica, for in 108 autopsy cases studied by Morse1 in which Brunn's nests and cysts were found in the urinary tract, more than 40% showed no inflammatory exudate. Moreover, fibrosis is considered to be part of the inflammatory reaction in ureteritis cystica.1 Our patient's tendency to form a left ureteral kink best explains the failure of surgery to relieve completely the ureteropelvic obstruction. This failure indicates that his original obstructive urographic deformity was not caused by ureteritis cystica alone but to some condition still present, possibly an excessively mobile left kidney which tends to settle into a position causing a kink at the ureteropelvic junction. Perhaps at operation, the kidney moved or was moved to a position not likely to produce a ureteral kink but after operation moved back to its ureter-kinking position, though with less obstruction at the kinked region as the result of removal of the ureteritis cystica. Since the flank pain which had been present since 1975 disappeared postoperatively, one might postulate that the ureteritis cystica had been present since then, along with the kinked ureter. We believe a kink could have served as the chronic irritant which produced the ureteritis cystica. Why the Brunn's nests did not develop into larger cysts is not entirely clear, but if the ureteritis cystica developed at the kink, it is possible the kinking caused a localized pressure preventing the Brunn's nests from expanding into the lumen as cysts. Such confinement may have produced fibrosis by acting as a chronic intramural irritant.
SUMMARY

In our case of ureteritis cystica, the ureteral obstruction was produced by localized fibrosis surrounding a cluster of Brunn's nests and small cysts. This is in contrast to the reported clinically diagnosed cases with
tFrom the Department of Pathology, Veterans Administration Medical Center, and Louisiana State University Medical Outer, New Orleans. Reprint requests to VA Medical Center. 1601 Perdido St. New Orleans, La 70146 (Dr. Sarma).

We have described a 58-year-old man in whom an unusually fibroblastic ureteritis cystica at the ureteropelvic junction caused partial obstruction and ureteral colic. Local resection resulted in relief of renal pain and improved urinary flow through the ureter. Explanations for the unusual nature of the lesion and only partial relief of obstruction after surgical resection are proposed.

Reprinted from the Southern Medical Journal, Journal of the Southern Medical Association, Volume 73, Number 7, July 1980, Pages 942-944, Copyright 1980 by Southern Medical Association, Birmingham, Alabama

FIGURE 1. Retrograde pyelogram shows dilated left (L) renal pelvis and calyces and obstruction at ureteropelvic junction.

FIGURE 2. Photomicrograph of ureteropelvic junction shows partial obstruction of lumen (L) by fibroblastic mass containing Brunn's epithelial nests. (H & E, original magnification x 24)

References
1. Morse HD: The etiology and pathology of pvelitis cystica, ureteritis cvstica arid cystitis cvstica. Am J Pathol 4:33-50, 19^8 2. McNultv M : Pyeloureteritis cystica. a review. Br] Radtol 30 :648-652, 1 957 3. Patch FS: Pyelitis, ureteritis, and cystitis cvstica. N Enei JIMed 220:979-987, 1939 '

Sarma DP, Lunseth JH (1980): Ureteritis cystica causing obstruction. South Med J 73:942-944.
PMID: 7384861 [PubMed - indexed for MEDLINE]