0022-5347/90/1441-0127$02 00/0 THE JOURNP,_L OF UROLOGY
Vol. 144, July Printed in U.S.A.
Copyright© 1990 by AMERICAN UROLOGICAL ASSOCIATION, INC.
Case Reports EOSINOPHILIC URETERITIS ASSOCIATED WITH EOSINOPHILIC CHOLANGITIS: A CASE REPORT McKAY L. PLATT, VICTOR J. KIESLING, JR. AND JOHN A. VACCARO From the Department of Surgery (Urology Service), Madigan Army Medical Center, Tacoma, Washington
ABSTRACT
Eosinophilic infiltrate of the urinary tract is rare. We report on a patient with eosinophilic cholangitis who later had eosinophilic ureteritis. This case emphasizes the potential serious nature of eosinophilic ureteritis, which often presents with complete ureteral obstruction. The literature pertaining to eosinophilic ureteritis and eosinophilic biliary disease is reviewed. (J. Ural., 144: 127129, 1990) Nonspecific inflammatory lesions of the ureter are rare. Less than 2 dozen cases have been reported in the literature. In 6 cases an eosinophilic infiltrate was described. A similar eosin ophilic infiltrate of the bladder has been termed eosinophilic cystitis and approximately 40 cases have been reported in the literature. We report a case of eosinophilic ureteritis associated with eosinophilic biliary disease.
The gallbladder contained no stones grossly. Postoperatively, the patient was managed with t-tube drainage of the common bile duct but later required a Roux-en-y operation in 1977 with common bile duct stenting because of common bile duct stenos1s.
DISCUSSION
A variety of apparently unrelated conditions lead to the CASE REPORT proliferation of eosinophils in the blood and in various tissues. A 56-year-old Filipino woman presented with several epi- Peripheral eosinophilia is seen in allergic disorders, autoimsodes of gross hematuria and acute renal failure (creatinine mune disorders, skin diseases, parasitic infections and certain 12.5, normal levels 1.0 to 1.7 for men and 0.8 to 1.4 for women). diseases of the hematopoietic system. Tissue eosinophilia is Urological evaluation showed bilateral ureteral obstruction and less common but has been described in eosinophilic granuloma a filling defect in the left distal third of the ureter. Cystoscopy of the bone (histiocytosis X). The gastrointestinal and genitoshowed patchy erythema, which was biopsied. The right ureter urinary tracts occasionally are involved with eosinophilic infilwas stented and the renal failure improved. At exploratory trates. Many reports exist of eosinophilic gastroenteritis (more laparotomy markedly thickened and inflamed middle and distal than 100 cases) and eosinophilic cystitis (42). Eosinophilic third ureters were found bilaterally. A sclerotic inflammatory infiltrate of the gallbladder with associated gall stones has been mass around the left distal third of the ureter was excised and described only 4 times in the literature. 1 ' 2 To our knowledge, eosinophilic cholangitis has never been described previously. left ureteroneocystostomy was performed. The peripheral eosinophil count was normal. Urine cultures, Eosinophilic ureteritis also is rare, with only 6 reported including cultures for acid-fast bacilli, stool for ova and para- cases. 3 - 8 Eosinophilic prostatitis 9 and eosinophilic orchitis 10 sites (hookworm, strongyloides, toxocariasis, trichuriasis and also have been described (more than 100 cases each). Of all cases of inflammatory ureteritis only 6 describe an schistosomiasis), allergy panel (food, tree, grass, weed, environmental and mold allergens), antinuclear antibodies, rheumatoid eosinophilic infiltrate (see table). 3 - 8 The intensity of the eosinfactor, quantitative immunoglobulins, cryoglobulins and serum ophilia varied from mild to dense and was associated with complement were all normal. Subsequent management of the peripheral eosinophilia in 3 cases. 5 · 7 Two cases were associated eosinophilic ureteritis has required continuous bilateral ure- with eosinophilic cystitis. 6 • 7 The etiology of eosinophilic cystitis and eosinophilic ureteritis. is not known but the 2 disorders teral stenting. The bladder biopsies showed chronic inflammation composed probably are related. Six cases of eosinophilic cystitis describe of lymphocytes and a mild eosinophilic infiltrate (fig. l, A). an associated history of allergy. Hellstrom and associates reThe excised distal third ureteral mass showed fibrosis with an ported 16 cases of eosinophilic cystitis associated with a prior operation or other local injury. 11 In addition, parasitic infestaintense eosinophilic infiltrate (fig. 1, B ). The patient had been hospitalized 14 years previously with tion has been suggested as a possible etiology but with little symptoms of common bile duct obstruction. Exploratory lapa- support. 12 The diagnosis of eosinophilic cystitis and eosinophilic urerotomy revealed an acutely inflamed gallbladder and cystic duct. Cholecystectomy and common bile duct exploration re- teritis is difficult or impossible to make without tissue biopsy. vealed severe inflammation and occlusion of the common duct The peripheral eosinophil count may be helpful if elevated but lumen, with inflammation with eosinophils and fibrosis (fig. 2). it is normal in approximately 65% of the reported series. The cystoscopic and radiographic findings also are nonspecific. Radiographic evaluation in eosinophilic cystitis may show filling Accepted for publication December 6, 1989. The opinions or assertions contained herein are the private views of defects of the bladder, a diminished bladder capacity or signs the authors and are not to be construed as official or as reflecting the of ureteral obstruction. Pathological examination of tissue reviews of the Department of the Army or the Department of Defense. mains the only means of diagnosis. 127
128
PLATT, KIESLING AND VACCARO
FIG. 1. A, bladder mucosa! biopsy demonstrates eosinophilic infiltrate and lymphocytes. H & E, reduced from X200. B , distal ureter shows intense eosinophilic infiltrate. H & E, reduced from XlOO.
FIG. 2. Bile duct with lymphocytic and eosinophilic infiltrates. Note cystic duct lumen above. H & E, reduced from X200.
129
EOSII\JOPI--!ILIC URETERJT'IS ASSOCIATED VVITH EOS!l"~OPHILIC Cr--l01-,Al¼GITi1~
Clinical features of eosinophilic ureteritis %
Ureteral Obstruction
Age-Sex
Presentation
Peripheral Eosinophilia
Felber' O'Flynn and Sandrey4 Hellstrom and associates'
70-F 40-F 21-M
Rt. flank pain Gross hematuria Lt. flank pain
? ?
Yes Yes Yes
Uyama and associates 6 Okafo and associates 7
37-M 72-M
6 6
Yes Yes
Mitas and Thompson 8
33-F
Lt. flank pain Rt. flank pain, irritative voiding Back pain, fever, irritative voiding
59
Yes
Reference
Oral steroids have been used successfully in the treatment of eosinophilic cystitis and eosinophilic ureteritis 7 • 13 but no one has reported reversal of ureteral obstruction with steroids. Unlike our patient, ureteral stenting was unsuccessful in the only case in which it was tried. 7 In a majority of the reported cases of eosinophilic ureteritis and in a number of cases of eosinophilic cystitis medical management failed and an open operation was required, including ureterectomy, nephroureterectomy and cystectomy with diversion. 3 • 5 • 7 • 14 Eosinophilic ureteritis is rare but it should be considered in the differential diagnosis of idiopathic ureteral obstruction, especially in patients with eosinophilic cystitis who have ureteral obstruction. Eosinophilic biliary disease also is rare. Its association with eosinophilic urothelial disease may be merely coincidental. Although neither disease has been associated with malignant degeneration both may cause significant morbidity.
REFERENCES
1. Kerstein, M. D., Sheahan, D. G., Gudjonsson, B. and Lewis, J.:
Eosinophilic cholecystitis. Amer. J. Gastroenterol., 66: 349, 1976. 2. Fox, H. and Mainwaring, A. R.: Eosinophilic infiltration of the gallbladder. Gastroenterology, 63: 1049, 1972. 3. Felber, E.: Granuloma of the ureter. J. Urol., 67: 152, 1952.
Treatment Nephroureterectomy Partial ureterectomy Lt. pyeloplasty, partia! ureterectomy Partial ureterectomy Partial cystectomy, partial ureterectomy Nephroureterectomy
Associated Eosinophilic Cystitis No No No
No Yes Yes
4. O'Flynn, W. R. and Sandrey, J. G.: Non-specific granulomata of the ureter and bladder. Brit. J. Urol., 35: 267, 1963. 5. Hellstrom, H. R., Davis, B. K, Shonnard, J. W. and MacPherson, T. A.: Eosinophilic pyeloureteritis: report of a case. J. Urol., 122: 833, 1979. 6. Uyama, T., Moriwaki, S., Aga, Y. and Yamamoto, A.: Eosinophilic ureteritis? Regional ureteritis with marked infiltration of eosinophils. Urology, 18: 615, 1981. 7. Okafo, B. A., Jones, H. W., Dow, D. and Kiruluta, H. G.: Eosinophilic cystitis: pleomorphic manifestations. Canad. J. Surg., 28: 17, 1985. 8. Mitas, J. A., II and Thompson, T.: Ureteral involvement complicating eosinophilic cystitis. Urology, 26: 67, 1985. 9. Redman, J. F. and Downs, R. A.: Simple eosinophilic granulomatous prostatitis. J. Urol., 132: 358, 1984. 10. Mostofi, F. K. and Price, E. B., Jr.: Tumors of the male genital system. Tumor-like conditions of the testis. In: Atlas of Tumor Pathology. Washington, D. C.: Armed Forces Institute of Pathology, 2nd series, fasc. 8, pp. 137-139, 1973. 11. Hellstrom, H. R., Davis, B. K. and Shonnard, J. W.: Eosinophilic cystitis. A study of 16 cases. Amer. J. Clin. Path., 72: 777, 1979. 12. Perlmutter, A. D., Edlow, J. B. and Kevy, S. W.: Toxocara antibodies in eosinophilic cystitis. J. Ped., 73: 340, 1968. 13. Kessler, W. 0., Clark, P. L. and Kaplan, G. W.: Eosinophilic cystitis. Urology, 6: 499, 1975. 14. Sidh, S. M., Smith, S. P., Silber, S. B. and Young, J. D., Jr.: Eosinophilic cystitis: advanced disease requiring surgical intervention. Urology, 15: 23, 1980.
Vol. 144, July Printed in U.S.A.
Copyright© 1990 by AMERICAN UROLOGICAL ASSOCIATION, INC.
Case Reports EOSINOPHILIC URETERITIS ASSOCIATED WITH EOSINOPHILIC CHOLANGITIS: A CASE REPORT McKAY L. PLATT, VICTOR J. KIESLING, JR. AND JOHN A. VACCARO From the Department of Surgery (Urology Service), Madigan Army Medical Center, Tacoma, Washington
ABSTRACT
Eosinophilic infiltrate of the urinary tract is rare. We report on a patient with eosinophilic cholangitis who later had eosinophilic ureteritis. This case emphasizes the potential serious nature of eosinophilic ureteritis, which often presents with complete ureteral obstruction. The literature pertaining to eosinophilic ureteritis and eosinophilic biliary disease is reviewed. (J. Ural., 144: 127129, 1990) Nonspecific inflammatory lesions of the ureter are rare. Less than 2 dozen cases have been reported in the literature. In 6 cases an eosinophilic infiltrate was described. A similar eosin ophilic infiltrate of the bladder has been termed eosinophilic cystitis and approximately 40 cases have been reported in the literature. We report a case of eosinophilic ureteritis associated with eosinophilic biliary disease.
The gallbladder contained no stones grossly. Postoperatively, the patient was managed with t-tube drainage of the common bile duct but later required a Roux-en-y operation in 1977 with common bile duct stenting because of common bile duct stenos1s.
DISCUSSION
A variety of apparently unrelated conditions lead to the CASE REPORT proliferation of eosinophils in the blood and in various tissues. A 56-year-old Filipino woman presented with several epi- Peripheral eosinophilia is seen in allergic disorders, autoimsodes of gross hematuria and acute renal failure (creatinine mune disorders, skin diseases, parasitic infections and certain 12.5, normal levels 1.0 to 1.7 for men and 0.8 to 1.4 for women). diseases of the hematopoietic system. Tissue eosinophilia is Urological evaluation showed bilateral ureteral obstruction and less common but has been described in eosinophilic granuloma a filling defect in the left distal third of the ureter. Cystoscopy of the bone (histiocytosis X). The gastrointestinal and genitoshowed patchy erythema, which was biopsied. The right ureter urinary tracts occasionally are involved with eosinophilic infilwas stented and the renal failure improved. At exploratory trates. Many reports exist of eosinophilic gastroenteritis (more laparotomy markedly thickened and inflamed middle and distal than 100 cases) and eosinophilic cystitis (42). Eosinophilic third ureters were found bilaterally. A sclerotic inflammatory infiltrate of the gallbladder with associated gall stones has been mass around the left distal third of the ureter was excised and described only 4 times in the literature. 1 ' 2 To our knowledge, eosinophilic cholangitis has never been described previously. left ureteroneocystostomy was performed. The peripheral eosinophil count was normal. Urine cultures, Eosinophilic ureteritis also is rare, with only 6 reported including cultures for acid-fast bacilli, stool for ova and para- cases. 3 - 8 Eosinophilic prostatitis 9 and eosinophilic orchitis 10 sites (hookworm, strongyloides, toxocariasis, trichuriasis and also have been described (more than 100 cases each). Of all cases of inflammatory ureteritis only 6 describe an schistosomiasis), allergy panel (food, tree, grass, weed, environmental and mold allergens), antinuclear antibodies, rheumatoid eosinophilic infiltrate (see table). 3 - 8 The intensity of the eosinfactor, quantitative immunoglobulins, cryoglobulins and serum ophilia varied from mild to dense and was associated with complement were all normal. Subsequent management of the peripheral eosinophilia in 3 cases. 5 · 7 Two cases were associated eosinophilic ureteritis has required continuous bilateral ure- with eosinophilic cystitis. 6 • 7 The etiology of eosinophilic cystitis and eosinophilic ureteritis. is not known but the 2 disorders teral stenting. The bladder biopsies showed chronic inflammation composed probably are related. Six cases of eosinophilic cystitis describe of lymphocytes and a mild eosinophilic infiltrate (fig. l, A). an associated history of allergy. Hellstrom and associates reThe excised distal third ureteral mass showed fibrosis with an ported 16 cases of eosinophilic cystitis associated with a prior operation or other local injury. 11 In addition, parasitic infestaintense eosinophilic infiltrate (fig. 1, B ). The patient had been hospitalized 14 years previously with tion has been suggested as a possible etiology but with little symptoms of common bile duct obstruction. Exploratory lapa- support. 12 The diagnosis of eosinophilic cystitis and eosinophilic urerotomy revealed an acutely inflamed gallbladder and cystic duct. Cholecystectomy and common bile duct exploration re- teritis is difficult or impossible to make without tissue biopsy. vealed severe inflammation and occlusion of the common duct The peripheral eosinophil count may be helpful if elevated but lumen, with inflammation with eosinophils and fibrosis (fig. 2). it is normal in approximately 65% of the reported series. The cystoscopic and radiographic findings also are nonspecific. Radiographic evaluation in eosinophilic cystitis may show filling Accepted for publication December 6, 1989. The opinions or assertions contained herein are the private views of defects of the bladder, a diminished bladder capacity or signs the authors and are not to be construed as official or as reflecting the of ureteral obstruction. Pathological examination of tissue reviews of the Department of the Army or the Department of Defense. mains the only means of diagnosis. 127
128
PLATT, KIESLING AND VACCARO
FIG. 1. A, bladder mucosa! biopsy demonstrates eosinophilic infiltrate and lymphocytes. H & E, reduced from X200. B , distal ureter shows intense eosinophilic infiltrate. H & E, reduced from XlOO.
FIG. 2. Bile duct with lymphocytic and eosinophilic infiltrates. Note cystic duct lumen above. H & E, reduced from X200.
129
EOSII\JOPI--!ILIC URETERJT'IS ASSOCIATED VVITH EOS!l"~OPHILIC Cr--l01-,Al¼GITi1~
Clinical features of eosinophilic ureteritis %
Ureteral Obstruction
Age-Sex
Presentation
Peripheral Eosinophilia
Felber' O'Flynn and Sandrey4 Hellstrom and associates'
70-F 40-F 21-M
Rt. flank pain Gross hematuria Lt. flank pain
? ?
Yes Yes Yes
Uyama and associates 6 Okafo and associates 7
37-M 72-M
6 6
Yes Yes
Mitas and Thompson 8
33-F
Lt. flank pain Rt. flank pain, irritative voiding Back pain, fever, irritative voiding
59
Yes
Reference
Oral steroids have been used successfully in the treatment of eosinophilic cystitis and eosinophilic ureteritis 7 • 13 but no one has reported reversal of ureteral obstruction with steroids. Unlike our patient, ureteral stenting was unsuccessful in the only case in which it was tried. 7 In a majority of the reported cases of eosinophilic ureteritis and in a number of cases of eosinophilic cystitis medical management failed and an open operation was required, including ureterectomy, nephroureterectomy and cystectomy with diversion. 3 • 5 • 7 • 14 Eosinophilic ureteritis is rare but it should be considered in the differential diagnosis of idiopathic ureteral obstruction, especially in patients with eosinophilic cystitis who have ureteral obstruction. Eosinophilic biliary disease also is rare. Its association with eosinophilic urothelial disease may be merely coincidental. Although neither disease has been associated with malignant degeneration both may cause significant morbidity.
REFERENCES
1. Kerstein, M. D., Sheahan, D. G., Gudjonsson, B. and Lewis, J.:
Eosinophilic cholecystitis. Amer. J. Gastroenterol., 66: 349, 1976. 2. Fox, H. and Mainwaring, A. R.: Eosinophilic infiltration of the gallbladder. Gastroenterology, 63: 1049, 1972. 3. Felber, E.: Granuloma of the ureter. J. Urol., 67: 152, 1952.
Treatment Nephroureterectomy Partial ureterectomy Lt. pyeloplasty, partia! ureterectomy Partial ureterectomy Partial cystectomy, partial ureterectomy Nephroureterectomy
Associated Eosinophilic Cystitis No No No
No Yes Yes
4. O'Flynn, W. R. and Sandrey, J. G.: Non-specific granulomata of the ureter and bladder. Brit. J. Urol., 35: 267, 1963. 5. Hellstrom, H. R., Davis, B. K, Shonnard, J. W. and MacPherson, T. A.: Eosinophilic pyeloureteritis: report of a case. J. Urol., 122: 833, 1979. 6. Uyama, T., Moriwaki, S., Aga, Y. and Yamamoto, A.: Eosinophilic ureteritis? Regional ureteritis with marked infiltration of eosinophils. Urology, 18: 615, 1981. 7. Okafo, B. A., Jones, H. W., Dow, D. and Kiruluta, H. G.: Eosinophilic cystitis: pleomorphic manifestations. Canad. J. Surg., 28: 17, 1985. 8. Mitas, J. A., II and Thompson, T.: Ureteral involvement complicating eosinophilic cystitis. Urology, 26: 67, 1985. 9. Redman, J. F. and Downs, R. A.: Simple eosinophilic granulomatous prostatitis. J. Urol., 132: 358, 1984. 10. Mostofi, F. K. and Price, E. B., Jr.: Tumors of the male genital system. Tumor-like conditions of the testis. In: Atlas of Tumor Pathology. Washington, D. C.: Armed Forces Institute of Pathology, 2nd series, fasc. 8, pp. 137-139, 1973. 11. Hellstrom, H. R., Davis, B. K. and Shonnard, J. W.: Eosinophilic cystitis. A study of 16 cases. Amer. J. Clin. Path., 72: 777, 1979. 12. Perlmutter, A. D., Edlow, J. B. and Kevy, S. W.: Toxocara antibodies in eosinophilic cystitis. J. Ped., 73: 340, 1968. 13. Kessler, W. 0., Clark, P. L. and Kaplan, G. W.: Eosinophilic cystitis. Urology, 6: 499, 1975. 14. Sidh, S. M., Smith, S. P., Silber, S. B. and Young, J. D., Jr.: Eosinophilic cystitis: advanced disease requiring surgical intervention. Urology, 15: 23, 1980.
