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Xanthogranulomatous Pyelonephritis in a Renal Allograft: A Case Report
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0022-5347/89/1414-0926$02.00/0 Vol. 141, April
THE JOURNAL OF UROLOGY
Printed in U.S.A.
Copyright © 1989 by The Williams & Wilkins Co.
XANTHOGRANULOMATOUS PYELONEPHRITIS IN A RENAL ALLOGRAFT: A CASE REPORT BERNARD F. JONES, RANJIT S. NANRA, ALEXANDER B. F. GRANT, NICHOLAS W. FERGUSON AND KEVIN H. WHITE From the Departments of Nephrology, Urology and Pathology, Royal Newcastle Hospital, Newcastle, New South Wales, Australia
ABSTRACT
We report a case of xanthogranulomatous pyelonephritis in a renal allograft. The kidney was not removed and there was an initial response to antibiotic therapy, with amelioration of toxicity and improvement in renal function. However, the kidney failed 10 months later in association with histological changes of chronic rejection. (J. Urol., 141: 926-927, 1989) Xanthogranulomatous pyelonephritis is an unusual chronic inflammatory disease of the kidney characterized by focal or diffuse replacement of renal parenchyma by granulomatous tissue containing lipid-filled histiocytes. 1 Women are more commonly affected than men. The disorder frequently is associated with obstructive lesions of the urinary tract and an association with diabetes mellitus has been described. 2 Bacterial cultures of the urine usually are positive for either Escherichia coli or Proteus mirabilis. The disorder rarely is bilateral and nephrectomy is the recommended treatment. 1• 3 To our knowledge this disease has not been described in a renal allograft. CASE REPORT
A 57-year-old woman was hospitalized on July 28, 1984 with a 3-day history of anorexia, myalgia, chills and diarrhea. In 1974 she had received a cadaveric renal allograft for renal failure due to malignant hypertension. In 1978 diabetes mellitus developed, which was treated with tolbutamide. Between the transplantation and the current hospitalization regular urine cultures had identified 6 urinary infections with various organisms. The last was January 1983 and urine culture 1 month before hospitalization was sterile. Serum creatinine had been stable at approximately 1.2 mg.jdl. (normal 0.07 to 1.1). At hospitalization the temperature was 38.5C. There was tenderness over the renal transplant. Urinalysis was positive for protein, blood and nitrites. Immunosuppressive therapy consisted of 100 mg. azathioprine and 10 mg. prednisolone daily. Serum creatinine was 5.8 mg./dl., white cell count 11,900/ mm. 3 (normal 4,000 to 10,000) and erythrocyte sedimentation rate 108 mm. per hour (normal3 to 9). The patient was treated with floxacillin and gentamicin. Blood cultures at hospitalization yielded E. coli, and floxacillin was ceased. Urine was examined about 12 hours after antibiotics were started, and contained 40 X 103 leukocytes per mm. 3 (normal less than 10 X 103 /mm. 3 ) and 50 X 103 erythrocytes per mm. 3 (normal less than 10 X 103/mm. 3 ) but no growth. A transplant ultrasound and perfusion scan were normal. Abdominal computerized tomography (CT) showed an ill-defmed low density mass approximately 1 X 2 em. in the kidney and there were other smaller areas of low attenuation within the renal parenchyma. At exploration the kidney was diffusely enlarged, with multiple small yellowish areas visible. The largest of these was biopsied. Frozen section examination showed xanthogranulomatous pyelonephritis, which was confirmed later on paraffin sections of frozen tissue (see figure). Biopsy taken from renal cortex, which appeared normal macroscopically, showed acute interstitial nephritis with numerous polymorphs. There was no Accepted for publication October 17, 1988.
histological evidence of rejection. E. coli was cultured from renal biopsy tissue. The kidney was not removed and a gallium scan 12 days postoperatively showed avid uptake of gallium in the kidney. When fever was slow to resolve and because of concern about nephrotoxicity gentamicin was replaced after 1 week by trimethoprim -sulfamethoxazole and then cephalosporins. The patient improved progressively. When she was afebrile she was discharged 1 month after hospitalization on trimethoprimsulfamethoxazole. Serum creatinine was 1.8 mg.jdl. Azathioprine had been reduced temporarily to 25 mg. daily because of leukopenia. Three weeks later serum creatinine began to increase and this was unaffected by a lower dose oftrimethoprimsulfamethoxazole. A repeat gallium scan showed only a small amount of gallium uptake by the kidney, and a repeat abdominal CT scan showed reduced size of the renal mass and otherwise normal renal parenchyma. A renal perfusion scan showed reduced perfusion compared to the earlier scan. The patient remained afebrile and urine remained sterile but renal failure progressed. When serum creatinine was 4.5 mg./dl. a renal biopsy was performed, which showed chronic rejection. Soon thereafter, trimethoprim-sulfamethoxazole was replaced with cephalexin, which was replaced later with amoxicillin. Amoxicillin was continued until February 1985 when the serum creatinine was 6.9 mg.jdl. Hemodialysis was started on May 24. The patient became progressively debilitated after Pseudomonas septicemia and gastrointestinal hemorrhage, and she died on June 27. Permission for an autopsy was not granted. DISCUSSION
A firm diagnosis of xanthogranulomatous pyelonephritis can be made in this case based on the typical findings at operation and histology results. This disease has not been reported previously in a renal allograft, although it has occurred in the native kidney of a renal allograft recipient. 4 The unusual features were the short clinical history, absence of an obstructive lesion and chronic urinary infection. 1 Failure to culture E. coli from the urine despite positive culture of renal tissue may have been due to antibiotics given before the urine was cultured. Following antibiotic therapy fever and systemic symptoms abated, there was a significant improvement in renal function and abdominal CT findings, and gallium uptake by the kidney decreased. Thus, despite the adverse influence of immunosuppressive therapy, this case raises the possibility that xanthogranulomatous pyelonephritis may respond to antibiotics. There are also other reports of response to antibiotics. 5• 6 Although the kidney eventually failed approximately 1 year later, this seems to have been due to rejection rather than xanthogranulomatous pyelonephritis, since the continued absence of systemic toxicity, and the improved gallium scan and abdomi-
926
XANTHO GRANDLO MATOUS PYELOl\T EPHRITIS
927
pyeloneph ritis on a renal biopsy does not exclude its presence in the kidney. Whether rejection was triggered xanthogra nulomatou s pyeloneph ritis or the reduced dose of azathiopr ine is conjectural. Nephrecto my is the recommen ded treatmen t for xanthogra nulomato us pyeloneph ritis,'· 3 but in a renal allograft, when the result of this treatmen t is dialysis, a trial of antibiotic s may merit considera tion.
REFERENC ES l. Grainger, R. G., Longstaff, A. J. and Parsons, M.A.: Xanthogra n-
Renal biopsy demonstra tes characteris tic foamy histiocytes of xanthogranulo matous pyeloneph ritis (freezing artifact present). H & E, reduced from X400.
nal CT appearanc es suggest resolution of the inflamma tory process, while the change in renal perfusion and the altered renal histology were compatib le with the developm ent of rejection. However, failure to demonstr ate xanthogra nulomato us
ulomatous pyelonephr itis: a reappraisal . Lancet, 1: 1398, 1982. 2. Gammill, 8., Rabinowitz , J. G., Peace, R., Sorgen, S., Hurwitz, L. and Himmelfar b, E.: New thoughts concerning xanthogran ulomatous pyelonephr itis (X-P). Amer. J. Roentgen., 125: 154, 1975. 3. Xanthogra nulomatou s pyelonephr itis. Lancet, 2: 649, 1985. 4. Carson, C. C. and Weinerth, J. L.: Xanthogra nulomatou s pyelonephritis in renal transplant recipient. Urology, 23: 58, 1984. 5. Rasoulpou r, M., Banco, L., McKay, I. M., Hight, D. W. and Berman, M. M.: Treatment of focal xanthogran ulomatous pyelonephritis with antibiotics . J. Ped., 105: 423, 1984. 6. Kromer, E. P., Schaefer, R. M. and Riegger, A. J. G.: Successful medical treatment of xanthogran ulomatous pyeloneph ritis. Letter to the Editor. Clin. Nephrol., 29: 271, 1988.
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0022-5347/89/1414-0926$02.00/0 Vol. 141, April
THE JOURNAL OF UROLOGY
Printed in U.S.A.
Copyright © 1989 by The Williams & Wilkins Co.
XANTHOGRANULOMATOUS PYELONEPHRITIS IN A RENAL ALLOGRAFT: A CASE REPORT BERNARD F. JONES, RANJIT S. NANRA, ALEXANDER B. F. GRANT, NICHOLAS W. FERGUSON AND KEVIN H. WHITE From the Departments of Nephrology, Urology and Pathology, Royal Newcastle Hospital, Newcastle, New South Wales, Australia
ABSTRACT
We report a case of xanthogranulomatous pyelonephritis in a renal allograft. The kidney was not removed and there was an initial response to antibiotic therapy, with amelioration of toxicity and improvement in renal function. However, the kidney failed 10 months later in association with histological changes of chronic rejection. (J. Urol., 141: 926-927, 1989) Xanthogranulomatous pyelonephritis is an unusual chronic inflammatory disease of the kidney characterized by focal or diffuse replacement of renal parenchyma by granulomatous tissue containing lipid-filled histiocytes. 1 Women are more commonly affected than men. The disorder frequently is associated with obstructive lesions of the urinary tract and an association with diabetes mellitus has been described. 2 Bacterial cultures of the urine usually are positive for either Escherichia coli or Proteus mirabilis. The disorder rarely is bilateral and nephrectomy is the recommended treatment. 1• 3 To our knowledge this disease has not been described in a renal allograft. CASE REPORT
A 57-year-old woman was hospitalized on July 28, 1984 with a 3-day history of anorexia, myalgia, chills and diarrhea. In 1974 she had received a cadaveric renal allograft for renal failure due to malignant hypertension. In 1978 diabetes mellitus developed, which was treated with tolbutamide. Between the transplantation and the current hospitalization regular urine cultures had identified 6 urinary infections with various organisms. The last was January 1983 and urine culture 1 month before hospitalization was sterile. Serum creatinine had been stable at approximately 1.2 mg.jdl. (normal 0.07 to 1.1). At hospitalization the temperature was 38.5C. There was tenderness over the renal transplant. Urinalysis was positive for protein, blood and nitrites. Immunosuppressive therapy consisted of 100 mg. azathioprine and 10 mg. prednisolone daily. Serum creatinine was 5.8 mg./dl., white cell count 11,900/ mm. 3 (normal 4,000 to 10,000) and erythrocyte sedimentation rate 108 mm. per hour (normal3 to 9). The patient was treated with floxacillin and gentamicin. Blood cultures at hospitalization yielded E. coli, and floxacillin was ceased. Urine was examined about 12 hours after antibiotics were started, and contained 40 X 103 leukocytes per mm. 3 (normal less than 10 X 103 /mm. 3 ) and 50 X 103 erythrocytes per mm. 3 (normal less than 10 X 103/mm. 3 ) but no growth. A transplant ultrasound and perfusion scan were normal. Abdominal computerized tomography (CT) showed an ill-defmed low density mass approximately 1 X 2 em. in the kidney and there were other smaller areas of low attenuation within the renal parenchyma. At exploration the kidney was diffusely enlarged, with multiple small yellowish areas visible. The largest of these was biopsied. Frozen section examination showed xanthogranulomatous pyelonephritis, which was confirmed later on paraffin sections of frozen tissue (see figure). Biopsy taken from renal cortex, which appeared normal macroscopically, showed acute interstitial nephritis with numerous polymorphs. There was no Accepted for publication October 17, 1988.
histological evidence of rejection. E. coli was cultured from renal biopsy tissue. The kidney was not removed and a gallium scan 12 days postoperatively showed avid uptake of gallium in the kidney. When fever was slow to resolve and because of concern about nephrotoxicity gentamicin was replaced after 1 week by trimethoprim -sulfamethoxazole and then cephalosporins. The patient improved progressively. When she was afebrile she was discharged 1 month after hospitalization on trimethoprimsulfamethoxazole. Serum creatinine was 1.8 mg.jdl. Azathioprine had been reduced temporarily to 25 mg. daily because of leukopenia. Three weeks later serum creatinine began to increase and this was unaffected by a lower dose oftrimethoprimsulfamethoxazole. A repeat gallium scan showed only a small amount of gallium uptake by the kidney, and a repeat abdominal CT scan showed reduced size of the renal mass and otherwise normal renal parenchyma. A renal perfusion scan showed reduced perfusion compared to the earlier scan. The patient remained afebrile and urine remained sterile but renal failure progressed. When serum creatinine was 4.5 mg./dl. a renal biopsy was performed, which showed chronic rejection. Soon thereafter, trimethoprim-sulfamethoxazole was replaced with cephalexin, which was replaced later with amoxicillin. Amoxicillin was continued until February 1985 when the serum creatinine was 6.9 mg.jdl. Hemodialysis was started on May 24. The patient became progressively debilitated after Pseudomonas septicemia and gastrointestinal hemorrhage, and she died on June 27. Permission for an autopsy was not granted. DISCUSSION
A firm diagnosis of xanthogranulomatous pyelonephritis can be made in this case based on the typical findings at operation and histology results. This disease has not been reported previously in a renal allograft, although it has occurred in the native kidney of a renal allograft recipient. 4 The unusual features were the short clinical history, absence of an obstructive lesion and chronic urinary infection. 1 Failure to culture E. coli from the urine despite positive culture of renal tissue may have been due to antibiotics given before the urine was cultured. Following antibiotic therapy fever and systemic symptoms abated, there was a significant improvement in renal function and abdominal CT findings, and gallium uptake by the kidney decreased. Thus, despite the adverse influence of immunosuppressive therapy, this case raises the possibility that xanthogranulomatous pyelonephritis may respond to antibiotics. There are also other reports of response to antibiotics. 5• 6 Although the kidney eventually failed approximately 1 year later, this seems to have been due to rejection rather than xanthogranulomatous pyelonephritis, since the continued absence of systemic toxicity, and the improved gallium scan and abdomi-
926
XANTHO GRANDLO MATOUS PYELOl\T EPHRITIS
927
pyeloneph ritis on a renal biopsy does not exclude its presence in the kidney. Whether rejection was triggered xanthogra nulomatou s pyeloneph ritis or the reduced dose of azathiopr ine is conjectural. Nephrecto my is the recommen ded treatmen t for xanthogra nulomato us pyeloneph ritis,'· 3 but in a renal allograft, when the result of this treatmen t is dialysis, a trial of antibiotic s may merit considera tion.
REFERENC ES l. Grainger, R. G., Longstaff, A. J. and Parsons, M.A.: Xanthogra n-
Renal biopsy demonstra tes characteris tic foamy histiocytes of xanthogranulo matous pyeloneph ritis (freezing artifact present). H & E, reduced from X400.
nal CT appearanc es suggest resolution of the inflamma tory process, while the change in renal perfusion and the altered renal histology were compatib le with the developm ent of rejection. However, failure to demonstr ate xanthogra nulomato us
ulomatous pyelonephr itis: a reappraisal . Lancet, 1: 1398, 1982. 2. Gammill, 8., Rabinowitz , J. G., Peace, R., Sorgen, S., Hurwitz, L. and Himmelfar b, E.: New thoughts concerning xanthogran ulomatous pyelonephr itis (X-P). Amer. J. Roentgen., 125: 154, 1975. 3. Xanthogra nulomatou s pyelonephr itis. Lancet, 2: 649, 1985. 4. Carson, C. C. and Weinerth, J. L.: Xanthogra nulomatou s pyelonephritis in renal transplant recipient. Urology, 23: 58, 1984. 5. Rasoulpou r, M., Banco, L., McKay, I. M., Hight, D. W. and Berman, M. M.: Treatment of focal xanthogran ulomatous pyelonephritis with antibiotics . J. Ped., 105: 423, 1984. 6. Kromer, E. P., Schaefer, R. M. and Riegger, A. J. G.: Successful medical treatment of xanthogran ulomatous pyeloneph ritis. Letter to the Editor. Clin. Nephrol., 29: 271, 1988.