Hô pital Pellegrin-Tripode, Bordeaux, France. Key words: .... Elkhammas EA, Mutabagani KH, Sedmak DD, Tesi RJ, Henry can be problematic [6].
Nephrol Dial Transplant ( 1997) 12: 831–832
Nephrology Dialysis Transplantation
Case Report
Xanthogranulomatous pyelonephritis in the native kidney of a renal transplant recipient V. Boulanger1, P. Merville1, D. Morel1, J. L. Pariente2, C. Deminie` re3 and L. Potaux1 1Department of Nephrology and Renal Transplantation, 2Department of Urology, 3Department of Pathology, Hoˆpital Pellegrin-Tripode, Bordeaux, France
Key words: xanthogranulomatous pyelonephritis, renal transplantation, xanthoma cells
Introduction Xanthogranulomatous pyelonephritis [1 ] is a rare condition, which is rarer still in an allograft recipient. We had recently the opportunity to observe this pathology in such a recipient.
Case Report A 55-year-old renal allograft recipient was admitted with a 3-month history of chronic abdominal pain, malaise, and weight loss ( 2 kg) without fever. He had no record of urinary tract obstruction or calculi. In 1987 he had received a cadaveric renal allograft for renal failure which had appeared in 1979. The primary nephropathy was unknown, and a renal biopsy was not performed at that time because of the atrophic nature of his kidneys. Renal function recovered promptly after transplantation with an immunosuppressive regimen comprising cyclosporin A, azathioprine, and steroids. During the following years, he presented numerous urinary tract infections, with isolation of various organisms (mainly Escherichia coli and Staphylococcus aureus). Physical examination was normal. Laboratory results showed an important inflammatory syndrome with elevated levels of CRP ( 320 mg/l ), fibrinogen ( 7.8 g/l ), and accelerated erythrocyte sedimentation rate at 110 mm. White blood cell count was 10 000/mm3. Values of haemoglobin, liver enzymes, serum creatinine, and blood urea nitrogen were normal. Urine examination revealed leukocyturia without bacteriuria, and a protein level at 1.0 g per day. Haematuria was also present and morphological study Correspondence and oprint requests to: Dr Pierre Merville, Service de Ne´phrologie et Transplantation Re´ nale, Hopital PellegrinTripode, Place Ame´ lie Raba-Le´on, 33076, Bordeaux Cedex, France.
showed that 90% of the erythrocytes were distorted, without any additional abnormality. Abdominal radiography did not show any calcifications. Ultrasound examination discovered an enlarged left native kidney with a dilated collecting system, whereas vascularization and echogenicity of the allograft were unremarkable. As indicated in Figure 1, an abdominal CT scan confirmed mild hydronephrosis of the left native kidney, which appeared enlarged and hyperdense with regular limits and a substantial perirenal hypodense infiltration. These findings suggested lymphoma or urothelial tumour. A left-sided nephroureterectomy was performed. The kidney was macroscopically pale and surrounded with indurated fat tissue. Cross-sections of the organ displayed multiple yellowish foci and a markedly dilated pyelum, filled with a purulent material. Histological examination diagnosed xanthogranulomatous pyelonephritis with a typical pattern of histiocyte infiltration. The histiocytic nature of the infiltrative cells was confirmed by immunostaining using specific monoclonal antibody against CD68. The patient had an uncomplicated recovery after surgery and left the hospital with long-term antimicrobial therapy. Kidney
Fig. 1. Renal computerized tomography: enlarged left kidney with a dilated pyelum. The margin cannot be distinguished from the perirenal fat.
© 1997 European Renal Association–European Dialysis and Transplant Association
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graft function was unaected by this episode and we did not modify the immunosuppressive regimen.
Comment The incidence of xanthogranulomatous pyelonephritis is not known, a fact that probably reflects its rarity. In one series it was found in six of 1000 surgically proven cases of chronic pyelonephritis [2 ], and a review of the Mayo Clinic’s experience from 1918 to 1975 found only 26 cases of this condition [3 ]. The depicted case represents the unique patient we have observed out of more than 1400 renal recipients over the last 20 years in our own centre. The originality of this observation lies in the onset of xanthogranulomatous pyelonephritis in a renal transplant recipient. In this context, two situations can be clearly identified. First, xanthogranulomatous pyelonephritis may aect the native kidney, as in our case. The disease was first described in 1984 in a single native kidney following a living-donor kidney transplantation, and was surgically treated [4 ]. Bilateral native localization has been described and associated lesions, particularly renal carcinoma, are not infrequent [5 ]. Consequently we recommend surgical treatment as the optimal management of any solid mass of a native end-stage kidney in a transplanted patient. Second, xanthogranulomatous pyelonephritis may aect the kidney allograft itself. In this particular localization, dierential diagnosis with acute rejection can be problematic [6 ]. Non-contributive biopsy must be repeated, since the lesions can be focal. Massive
V. Boulanger et al.
unusual infiltration of CD68+ histiocytes, such as that demonstrated by the immunohistological staining we performed, may be considered as a reliable indication of the diagnosis. Surgical treatment is not mandatory and long-term antibiotic therapy has been successful in preserving graft function [7]. In conclusion, xanthogranulomatous pyelonephritis is a rare, but perhaps underestimated renal pathology which can aect kidney allograft recipients. Curative treatment consists in surgery, when the native kidney is aected, or antibiotic therapy, when the localization is in the allograft.
References 1. Stam F, van den Tillaar PLM, Falke ThHM, Gans ROB, ter Wee PM. Xanthogranulomatous pyelonephritis. Nephrol Dial Transplant 1995; 10: 1283–2405 2. Dussaix E, Wood C. Cytomegalovirus infection in pediatric liver recipients. A virological survey and prophylaxis with CMV immune globulin and early DHPG treatment. Transplantation 1989; 48: 272–274 3. Malek RS, Eloer JS. Xanthogranulomatous pyelonephritis: a critical analysis of 26 cases and of the literature. J Urol 1978; 119: 589–593 4. Carson CC, Weinerth JL. Xanthogranulomatous pyelonephritis in renal transplant recipient. Urology 1984; 23: 58–61 5. Akhtar M, Qunibi W. Bilateral xanthogranulomatous pyelonephritis involving native kidneys in a renal transplant recipient: association with renal cell carcinoma and amyloidosis. Am J Kidney Dis 1992; 20: 289–393 6. Jones BF, Nanra RS, Grant AB, Ferguson NW, White KH. Xanthogranulomatous pyelonephritis in a renal allograft: a case report. J Urol 1989; 141: 926–927 7. Elkhammas EA, Mutabagani KH, Sedmak DD, Tesi RJ, Henry ML, Ferguson RM. Xanthogranulomatous pyelonephritis in renal allografts: report of two cases. J Urol 1994; 151: 127–128 Received for publication: 4.11.96 Accepted: 14.11.96
