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CASE REPORT
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LATE RECURRENCE OF RENAL CHOLESTEATOMA AFTER15 YEARS
JENS KÖNIG, JÜRGEN PANNEK, RALPH KICKUTH, AND JOACHIM NOLDUS
ABSTRACTe report a late recurrence of a cholesteatoma of the left kidney after 15 years. Both the initial case and the
ecurrence were treated by endourologic and percutaneous approaches. UROLOGY 64: 808.e19–808.e20,004. © 2004 Elsevier Inc.
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holesteatoma of the urinary tract, first re-ported by Rosina1 in 1953 is an extremely un-
ommon disease. Only 90 cases have been reportedn published studies. Histologically, keratinizingesquamative squamous metaplasia of the urothe-ial cell layer is found.2 The most common site of
anifestation is the renal pelvis, but manifesta-ions in the ureter have been reported as well.3
CASE REPORT
In 1989, a 50-year-old woman presented to ourepartment with left flank pain and hematuria. Heredical history was uneventful, and the physical
xamination was normal. Urine examinationhowed no signs of acute infection but significantematuria; the urine culture was sterile. Her rou-ine blood tests were normal, with a serum creati-ine of 1.3 mg/dL. Urography and renal ultra-onography led to the diagnosis of a stone in theenal pelvis, which was removed using a percuta-eous approach. Histologic examination of the
ragments revealed a cholesteatoma of the renalelvis. The follow-up was uneventful, and no fur-her treatment was necessary. In December 2003,er flank pain and hematuria recurred. At thatime, urine examination confirmed keratin sheets,er serum creatinine was 1.5 mg/dL, and the otherlood tests were normal.Urography showed a large solid mass in the left
enal pelvis. Computed tomography and magnetic
rom the Departments of Urology and Radiology, Ruhr Univer-ität Bochum, Marienhospital Herne, Herne, Germany
Address for correspondence: Jens König, M.D., Department ofrology, Ruhr Universität Bochum, Marienhospital Herne, Wi-umer Strasse 8, Herne 44627, Germany
Submitted: March 1, 2004, accepted (with revisions): May 6,
t0042004 ELSEVIER INC.LL RIGHTS RESERVED
esonance imaging (Fig. 1) revealed a noninvasiverocess. Retrograde ureteropyelography (Fig. 2)onfirmed the diagnosis. Cystoscopy revealed aormal lower urinary tract, and left ureteropyelog-aphy did not show any other pathologic findings.or therapy, a percutaneous approach was used,nd a nephrostomy tube was inserted (Fig. 3). An-egrade pyeloureterography revealed no residualeratin material, and the tube was removed after 2ays. The histopathologic examination confirmedhe recurrence of the cholesteatoma.
COMMENT
Because renal cholesteatomas are rare, the stan-ard treatment has not been well defined. Untilecently, nephrectomy was reported as the treat-ent of choice. This aggressive procedure was per-
ormed because of the lack of information regard-ng the malignant potential and recurrence rate.owever, no malignant potential3 or any associa-
ion with squamous carcinomas has been de-cribed. In published studies, different nephron-paring procedures have been described, includingait and see strategies, pyelolithotomy,4 extracor-oral workbench surgery,5 partial nephrectomy,nd endourological approaches.6Important diagnostic features are keratin sheets
n urine cytology,7 the absence of malignant cells,nd radiologic defects on urography. The differen-ial diagnosis includes renal stones, tumors, in-ammatory and necrotic processes, and bloodlots, which must be excluded. The etiology of re-al cholesteatomas is unclear. Chronic infection,bstruction, and stones have been considered. Inur case, none of these factors were evaluated.The exact data regarding the recurrence rate of
his entity are very rare in published studies owing
0090-4295/04/$30.00doi:10.1016/j.urology.2004.05.008 808.e19
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o the small number of cases worldwide. It seemsbvious that cholesteatoma recurrence can occuruickly if the keratin matrix has not been com-letely removed.4 Taguchi et al.5 reported a case ofecurrence 7 years after pyeloscopic treatment.upovitch et al.8 described a case with a long-termecurrence-free follow-up of more than 56 years.otable in that long-term case was that supportive
itamin A therapy led to stabilization of the kera-inizing lesion. After discontinuing the therapy,he symptoms recurred. It is known that vitamin Aupplementation has significant value in other dis-
IGURE 1. Magnetic resonance imaging scan showingeterogeneous mass in left renal pelvis.
FIGURE 2. Retrograde ureteropyelography.
ases such as oral leukoplakia. J
08.e20
In our patient, the cholesteatoma recurred after5 years, although no supportive therapy had beensed, and a percutaneous approach led to quicknd satisfactory results.
REFERENCES
1. Rosina G: Two rare forms of chronic pyelitis: leukopla-ia with cholesteatoma and wide, isolated calcification of theenal pelvis. Osp Maggiore 41: 431–437, 1953.
2. Hertle L, and Androulakakis P: Keratinizing desquama-ive squamous metaplasia of the upper urinary tract–leukopla-ia–cholesteatoma. J Urol 127: 631–635, 1982.3. Weitzner S: Cholesteatoma of the calix. J Urol 108: 365–
67, 1972.4. Willis JS, Pollack H, and Curtis JA: Cholesteatoma of the
pper urinary tract. AJR Am J Roentgenol 136: 941–944,981.5. Taguchi Y, Kotha V, Tomka B, et al: Conserving
ephrons in cholesteatoma. J Urol 123: 258–260, 1980.6. Neerhut G, Politis G, Alpert L, et al: Cholesteatoma of
he renal pelvis: endoscopic management. J Urol 139: 1032–034, 1988.7. Gale GL, and Kerr WK: Cholesteatoma of the urinary
ract. J Urol 104(1): 71–72, 1970.8. Lupovitch A, Domzalski H, and Tippins R: Cholestea-
oma of the renal pelvis—a case with long term follow up.
FIGURE 3. Percutaneous approach.
Urol 140: 360–361, 1988.
UROLOGY 64 (4), 2004
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