0022-5347/79 /1226-0833$02.00/0
THE
Vol. 122, December
JOURNAL OF UROLOGY
Copyright© 1979 by The Williams & Wilkins Co.
Printed in U.S.A.
EOSINOPHILIC PYELOURETERITIS: REPORT OF A CASE H. RICHARD HELLSTROM,* BRIDGETT K. DAVIS, JOHN W. SHONNARD MACPHERSON
AND
TREVOR A.
From the Departments of Pathology, University of Pittsburgh School of Medicine and the Veterans Administration Hospital, Pittsburgh, Pennsylvania
ABSTRACT
A case is reported of ureteral obstruction that was owing to eosinophilic pyeloureteritis, a previously unrecorded entity. The microscopic findings of extensive fibrosis and a relatively mild eosinophilic infiltrate were similar to those found in a series of eosinophilic cystitis, which was reported recently from this laboratory. Also, local injury appears to initiate some examples of eosinophilic cystitis and in the present case there was a striking history of injury 1 month before the symptoms of ureteral obstruction. Eosinophilic pyeloureteritis has not been reported previously, although there have been a number of reports of eosinophilic cystitis. 1 Herein we describe a case of eosinophilic pyeloureteritis that occurred after injury and caused ureteral obstruction.
similarity, this lesion was designated as eosinophilic pyeloureteritis. Since the ureter and pelvis are similar histologically to the bladder it is likely that the morphologic changes and pathogenetic mechanisms of eosinophilic pyeloureteritis are similar to eosinophilic cystitis. However, the often dominant findings of fibrosis and scanty eosinophils in our recent series of 16 cases of eosinophilic cystitis differ from the usual understanding of eosinophilic cystitis derived from the accumulation of 21 case reports in the English literature. 1 In these cases acute changes, such as a prominent eosinophilic infiltration and marked mucosal edema, appear to be stressed. This emphasis is in part spurious, since a number of the earlier reports had brief microscopic descriptions that often were limited to noting the presence of eosinophils. Occasional reports did mention, but did not emphasize, findings such as fibrosis and a scanty eosinophilic infiltrate. Another reason for the apparent discrepancy in microscopic descriptions relates to the sequence of histologic changes that occurs after an acute attack. Eosinophilic cystitis and, presumably, eosinophilic pyeloureteritis are episodic. In the acute phase there is a prominent eosinophilic infiltrate and within several weeks eosinophils diminish and chronic inflammation and fibrosis supervene. Microscopic findings reflect the stage of the inflammatory process and whether prior attacks have occurred. Biopsies taken after repeated episodes and at some interval after the last attack, as tended to occur in our cases of eosinophilic cystitis, would demonstrate fibrosis and a relative paucity of eosinophils. In the present case an operation was performed for chronic ureteral obstruction and chronic microscopic changes were to be expected. When diagnosing eosinophilic cystitis and pyeloureteritis one should keep in mind that acute and chronic changes in varying combinations may be present. We regard muscle necrosis as an integral feature of this disorder and consider necrosis and its sequela, replacement fibrosis of muscle, as important diagnostic criteria. In our case of eosinophilic pyeloureteritis there was significant fibrous replacement of muscle, a finding that reinforces the diagnosis and relates this disorder to eosinophilic cystitis. Replacement fibrosis is diagnosed by the identification of muscle remnants in scar, and the trichrome stain is helpful in identifying these muscle fragments. Also, the presence and amount of eosinophils are better appreciated by the Giemsa stain. Our previous study of eosinophilic cystitis provides other information that is directly or indirectly relevant to eosinophilic pyeloureteritis or ureteritis. 1 Eosinophilic cystitis could be divided into 2 overlapping groups. The first group, which included patients from the earlier series of case reports in the English literature, had allergies and peripheral eosinophilia, and con-
CASE REPORT
B. J.M., a 21-year-old white man, was hospitalized because of recurrent left flank pain. He had had an accident aboard ship 17 months previously, while he was in the Navy. Barrels broke loose and pinned him against a 4-foot railing. The railing then gave way and he was washed overboard. He was "black and blue all over". A month later left flank pain developed and 2 months thereafter ureteropelvic junction repair was done for ureteral obstruction in a military hospital. No tissue was removed. The patient was free of symptoms for 8 months but during the last 6 months the left flank pain recurred. White blood counts were 9,000 per mm.a with 1 per cent eosinophils and 14,400 per mm.a with O per cent eosinophils. An excretory urogram showed a normal right kidney and faint nephrogram effect on the left side without any evidence of excretion. At cystoscopy a catheter was passed with some difficulty through the left ureteropelvic junction and was followed by an immediate hydronephrotic drip. Approximately 20 ml. dye were required to fill the pelvis and retrograde pyelography revealed a huge pyelonephrotic sac. The stenotic ureteropelvic segment was excised surgically and a ureteropelvic anastomosis was done. The surgical specimen consisted of a 3.5 cm. length of ureter and a 1.3 cm. attached portion of pelvis. The ureter was thickened, measuring up to 0.8 cm. in diameter, and the lumen was narrow. Microscopically, in the ureter and pelvis there was fibrosis of the mucosa, muscularis and perimuscular tissue (part A of figure). Fibrosis in the muscularis separated and replaced muscle bundles and in several areas of the ureter the muscularis was replaced completely by scar (part B of figure). There was focal mild chronic inflammation, which contained areas of scattered eosinophils (part C of figure). Focal mild ureteral mucosal edema also was present. DISCUSSION
The major microscopic changes present in the resected segment of the pelvis and ureter were fibrosis and a mild chronic inflammation that contained scattered eosinophils. Such changes were observed in a series of cases of eosinophilic cystitis recently reported from this laboratory1 and, because of this Accepted for publication February 9, 1979. * Requests for reprints: Laboratory Service, Veterans Administration Hospital, Irving Ave. and University Place, Syracuse, New York 13210. 833
834
HELLSTROM AND ASSOCIATES
A, histologic section of ureter shows fibrosis of mucosa, muscularis and perimuscular tissue. Muscle bundles are separated by fibrosis and some muscle bundles are small, suggesting loss of muscularis with replacement fibrosis. Trichrome, reduced from X32. B, histologic section of ureter with extensive fibrosis. Much of muscularis has been replaced by fibrosis and on right side there is complete loss of muscularis. Trichrome, reduced from X32. C, higher power photomicrograph shows replacement fibrosis of muscle and focal mild chronic inflammatory infiltrate that contains scattered eosinophils. Eosinophils cannot be identified in photomicrograph. H & E, reduced from X96.
sisted mainly of women and children. The second group, which included almost all patients in our previous series, was related to bladder injury, was unassociated with allergy or peripheral eosinophilia and usually occurred in older men. Injuries included bladder obstruction owing to benign prostatic hypertrophy, bladder carcinoma and surgical procedures. In our case of eosinophilic pyeloureteritis the striking history of shipboard injury 1 month before the development of flank pain provides another link to eosinophilic cystitis and is further evidence for the injury mechanism of eosinophilic urinary tract inflammations. The injury type of eosinophilic cystitis probably occurs fairly commonly. Of 17 cases of eosinophilic cystitis reported by Zeitlhofer and Bibus2 11 had associated bladder conditions, and Melicow and Uson 3 reported 147 examples of herald lesions, which appear to represent the injury type of eosinophilic cystitis. The injury type probably tends to be misdiagnosed. Eosinophilic cystitis might not be considered in older men with
bladder conditions as obstructive uropathy and in our previous series the clinical diagnosis was not suspected in any case. Also, in biopsies it is our experience that microscopists not alerted to the range of findings in eosinophilic cystitis invariably overlook the diagnosis. We suggest that eosinophilic pyeloureteritis and ureteritis be considered in the differential diagnosis of ureteral obstruction. A variety of traumatic insults to the ureter might initiate eosinophilic ureteritis and pyeloureteritis. REFERENCES 1. Hellstrom, H. R., Davis, B. K. and Shonnard, J. W.: Eosinophilic cystitis: a study of 16 cases. Amer. J. Clin. Path., in press. 2. Zeitlhofer, J. and Bibus, B.: Zur Klinik und Pathologie des eosinophilen Harblaseninfiltrates. Wein. Klein. Wochenschr., 79: 958, 1967. 3. Melicow, M. M. and Uson, A. C.: The "herald" lesion of the bladder: a lesion which portends the approach of cancer or inflammation from outside the bladder. J. Urol., 85: 543, 1961.
THE
Vol. 122, December
JOURNAL OF UROLOGY
Copyright© 1979 by The Williams & Wilkins Co.
Printed in U.S.A.
EOSINOPHILIC PYELOURETERITIS: REPORT OF A CASE H. RICHARD HELLSTROM,* BRIDGETT K. DAVIS, JOHN W. SHONNARD MACPHERSON
AND
TREVOR A.
From the Departments of Pathology, University of Pittsburgh School of Medicine and the Veterans Administration Hospital, Pittsburgh, Pennsylvania
ABSTRACT
A case is reported of ureteral obstruction that was owing to eosinophilic pyeloureteritis, a previously unrecorded entity. The microscopic findings of extensive fibrosis and a relatively mild eosinophilic infiltrate were similar to those found in a series of eosinophilic cystitis, which was reported recently from this laboratory. Also, local injury appears to initiate some examples of eosinophilic cystitis and in the present case there was a striking history of injury 1 month before the symptoms of ureteral obstruction. Eosinophilic pyeloureteritis has not been reported previously, although there have been a number of reports of eosinophilic cystitis. 1 Herein we describe a case of eosinophilic pyeloureteritis that occurred after injury and caused ureteral obstruction.
similarity, this lesion was designated as eosinophilic pyeloureteritis. Since the ureter and pelvis are similar histologically to the bladder it is likely that the morphologic changes and pathogenetic mechanisms of eosinophilic pyeloureteritis are similar to eosinophilic cystitis. However, the often dominant findings of fibrosis and scanty eosinophils in our recent series of 16 cases of eosinophilic cystitis differ from the usual understanding of eosinophilic cystitis derived from the accumulation of 21 case reports in the English literature. 1 In these cases acute changes, such as a prominent eosinophilic infiltration and marked mucosal edema, appear to be stressed. This emphasis is in part spurious, since a number of the earlier reports had brief microscopic descriptions that often were limited to noting the presence of eosinophils. Occasional reports did mention, but did not emphasize, findings such as fibrosis and a scanty eosinophilic infiltrate. Another reason for the apparent discrepancy in microscopic descriptions relates to the sequence of histologic changes that occurs after an acute attack. Eosinophilic cystitis and, presumably, eosinophilic pyeloureteritis are episodic. In the acute phase there is a prominent eosinophilic infiltrate and within several weeks eosinophils diminish and chronic inflammation and fibrosis supervene. Microscopic findings reflect the stage of the inflammatory process and whether prior attacks have occurred. Biopsies taken after repeated episodes and at some interval after the last attack, as tended to occur in our cases of eosinophilic cystitis, would demonstrate fibrosis and a relative paucity of eosinophils. In the present case an operation was performed for chronic ureteral obstruction and chronic microscopic changes were to be expected. When diagnosing eosinophilic cystitis and pyeloureteritis one should keep in mind that acute and chronic changes in varying combinations may be present. We regard muscle necrosis as an integral feature of this disorder and consider necrosis and its sequela, replacement fibrosis of muscle, as important diagnostic criteria. In our case of eosinophilic pyeloureteritis there was significant fibrous replacement of muscle, a finding that reinforces the diagnosis and relates this disorder to eosinophilic cystitis. Replacement fibrosis is diagnosed by the identification of muscle remnants in scar, and the trichrome stain is helpful in identifying these muscle fragments. Also, the presence and amount of eosinophils are better appreciated by the Giemsa stain. Our previous study of eosinophilic cystitis provides other information that is directly or indirectly relevant to eosinophilic pyeloureteritis or ureteritis. 1 Eosinophilic cystitis could be divided into 2 overlapping groups. The first group, which included patients from the earlier series of case reports in the English literature, had allergies and peripheral eosinophilia, and con-
CASE REPORT
B. J.M., a 21-year-old white man, was hospitalized because of recurrent left flank pain. He had had an accident aboard ship 17 months previously, while he was in the Navy. Barrels broke loose and pinned him against a 4-foot railing. The railing then gave way and he was washed overboard. He was "black and blue all over". A month later left flank pain developed and 2 months thereafter ureteropelvic junction repair was done for ureteral obstruction in a military hospital. No tissue was removed. The patient was free of symptoms for 8 months but during the last 6 months the left flank pain recurred. White blood counts were 9,000 per mm.a with 1 per cent eosinophils and 14,400 per mm.a with O per cent eosinophils. An excretory urogram showed a normal right kidney and faint nephrogram effect on the left side without any evidence of excretion. At cystoscopy a catheter was passed with some difficulty through the left ureteropelvic junction and was followed by an immediate hydronephrotic drip. Approximately 20 ml. dye were required to fill the pelvis and retrograde pyelography revealed a huge pyelonephrotic sac. The stenotic ureteropelvic segment was excised surgically and a ureteropelvic anastomosis was done. The surgical specimen consisted of a 3.5 cm. length of ureter and a 1.3 cm. attached portion of pelvis. The ureter was thickened, measuring up to 0.8 cm. in diameter, and the lumen was narrow. Microscopically, in the ureter and pelvis there was fibrosis of the mucosa, muscularis and perimuscular tissue (part A of figure). Fibrosis in the muscularis separated and replaced muscle bundles and in several areas of the ureter the muscularis was replaced completely by scar (part B of figure). There was focal mild chronic inflammation, which contained areas of scattered eosinophils (part C of figure). Focal mild ureteral mucosal edema also was present. DISCUSSION
The major microscopic changes present in the resected segment of the pelvis and ureter were fibrosis and a mild chronic inflammation that contained scattered eosinophils. Such changes were observed in a series of cases of eosinophilic cystitis recently reported from this laboratory1 and, because of this Accepted for publication February 9, 1979. * Requests for reprints: Laboratory Service, Veterans Administration Hospital, Irving Ave. and University Place, Syracuse, New York 13210. 833
834
HELLSTROM AND ASSOCIATES
A, histologic section of ureter shows fibrosis of mucosa, muscularis and perimuscular tissue. Muscle bundles are separated by fibrosis and some muscle bundles are small, suggesting loss of muscularis with replacement fibrosis. Trichrome, reduced from X32. B, histologic section of ureter with extensive fibrosis. Much of muscularis has been replaced by fibrosis and on right side there is complete loss of muscularis. Trichrome, reduced from X32. C, higher power photomicrograph shows replacement fibrosis of muscle and focal mild chronic inflammatory infiltrate that contains scattered eosinophils. Eosinophils cannot be identified in photomicrograph. H & E, reduced from X96.
sisted mainly of women and children. The second group, which included almost all patients in our previous series, was related to bladder injury, was unassociated with allergy or peripheral eosinophilia and usually occurred in older men. Injuries included bladder obstruction owing to benign prostatic hypertrophy, bladder carcinoma and surgical procedures. In our case of eosinophilic pyeloureteritis the striking history of shipboard injury 1 month before the development of flank pain provides another link to eosinophilic cystitis and is further evidence for the injury mechanism of eosinophilic urinary tract inflammations. The injury type of eosinophilic cystitis probably occurs fairly commonly. Of 17 cases of eosinophilic cystitis reported by Zeitlhofer and Bibus2 11 had associated bladder conditions, and Melicow and Uson 3 reported 147 examples of herald lesions, which appear to represent the injury type of eosinophilic cystitis. The injury type probably tends to be misdiagnosed. Eosinophilic cystitis might not be considered in older men with
bladder conditions as obstructive uropathy and in our previous series the clinical diagnosis was not suspected in any case. Also, in biopsies it is our experience that microscopists not alerted to the range of findings in eosinophilic cystitis invariably overlook the diagnosis. We suggest that eosinophilic pyeloureteritis and ureteritis be considered in the differential diagnosis of ureteral obstruction. A variety of traumatic insults to the ureter might initiate eosinophilic ureteritis and pyeloureteritis. REFERENCES 1. Hellstrom, H. R., Davis, B. K. and Shonnard, J. W.: Eosinophilic cystitis: a study of 16 cases. Amer. J. Clin. Path., in press. 2. Zeitlhofer, J. and Bibus, B.: Zur Klinik und Pathologie des eosinophilen Harblaseninfiltrates. Wein. Klein. Wochenschr., 79: 958, 1967. 3. Melicow, M. M. and Uson, A. C.: The "herald" lesion of the bladder: a lesion which portends the approach of cancer or inflammation from outside the bladder. J. Urol., 85: 543, 1961.
