Late recurrence of renal cholesteatoma after 15 years

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    Idemeexshhetinninsorenal pelvis, which was removed using a percuta-neous approach. Histologic examination of thefrapethhetimheblo

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    CASE REPORT

    ALLgments revealed a cholesteatoma of the renallvis. The follow-up was uneventful, and no fur-er treatment was necessary. In December 2003,r flank pain and hematuria recurred. At thate, urine examination confirmed keratin sheets,

    r serum creatinine was 1.5 mg/dL, and the otherod tests were normal.rography showed a large solid mass in the leftal pelvis. Computed tomography and magnetic

    tion with squamous carcinomas has been de-scribed. In published studies, different nephron-sparing procedures have been described, includingwait and see strategies, pyelolithotomy,4 extracor-poral workbench surgery,5 partial nephrectomy,and endourological approaches.6

    Important diagnostic features are keratin sheetsin urine cytology,7 the absence of malignant cells,and radiologic defects on urography. The differen-tial diagnosis includes renal stones, tumors, in-flammatory and necrotic processes, and bloodclots, which must be excluded. The etiology of re-nal cholesteatomas is unclear. Chronic infection,obstruction, and stones have been considered. In

    m the Departments of Urology and Radiology, Ruhr Univer-t Bochum, Marienhospital Herne, Herne, Germanyddress for correspondence: Jens Knig, M.D., Department ofLATE RECURRENCE OF RENA15 YE

    JENS KNIG, JRGEN PANNEK, RALP

    ABSTRreport a late recurrence of a cholesteatoma of the lurrence were treated by endourologic and percutan04. 2004 Elsevier Inc.

    holesteatoma of the urinary tract, first re-ported by Rosina1 in 1953 is an extremely un-

    mmon disease. Only 90 cases have been reportedpublished studies. Histologically, keratinizing

    squamative squamous metaplasia of the urothe-l cell layer is found.2 The most common site ofnifestation is the renal pelvis, but manifesta-ns in the ureter have been reported as well.3

    CASE REPORT

    n 1989, a 50-year-old woman presented to ourpartment with left flank pain and hematuria. Herdical history was uneventful, and the physical

    amination was normal. Urine examinationowed no signs of acute infection but significantmaturia; the urine culture was sterile. Her rou-e blood tests were normal, with a serum creati-e of 1.3 mg/dL. Urography and renal ultra-

    nography led to the diagnosis of a stone in theouT

    th

    ology, Ruhr Universitt Bochum, Marienhospital Herne, Wi-er Strasse 8, Herne 44627, Germanyubmitted: March 1, 2004, accepted (with revisions): May 6,4

    2004 ELSEVIER INC.RIGHTS RESERVEDCHOLESTEATOMA AFTERRS

    ICKUTH, AND JOACHIM NOLDUS

    Tidney after 15 years. Both the initial case and thes approaches. UROLOGY 64: 808.e19808.e20,

    onance imaging (Fig. 1) revealed a noninvasiveocess. Retrograde ureteropyelography (Fig. 2)nfirmed the diagnosis. Cystoscopy revealed armal lower urinary tract, and left ureteropyelog-hy did not show any other pathologic findings.r therapy, a percutaneous approach was used,d a nephrostomy tube was inserted (Fig. 3). An-rade pyeloureterography revealed no residualratin material, and the tube was removed after 2ys. The histopathologic examination confirmede recurrence of the cholesteatoma.

    COMMENT

    ecause renal cholesteatomas are rare, the stan-rd treatment has not been well defined. Untilently, nephrectomy was reported as the treat-nt of choice. This aggressive procedure was per-med because of the lack of information regard-r case, none of these factors were evaluated.he exact data regarding the recurrence rate of

    is 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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    FIGURE 1. Magnetic resonance imaging scan showinghe

    80the small number of cases worldwide. It seemsvious that cholesteatoma recurrence can occurickly if the keratin matrix has not been com-tely removed.4 Taguchi et al.5 reported a case ofurrence 7 years after pyeloscopic treatment.povitch et al.8 described a case with a long-termurrence-free follow-up of more than 56 years.table in that long-term case was that supportiveamin A therapy led to stabilization of the kera-izing lesion. After discontinuing the therapy,

    e symptoms recurred. It is known that vitamin Applementation has significant value in other dis-ses such as oral leukoplakia.

    terogeneous mass in left renal pelvis.

    FIGURE 2. Retrograde ureteropyelography.8.e20n our patient, the cholesteatoma recurred afteryears, although no supportive therapy had been

    ed, and a percutaneous approach led to quickd satisfactory results.

    REFERENCES

    . Rosina G: Two rare forms of chronic pyelitis: leukopla-with cholesteatoma and wide, isolated calcification of theal pelvis. Osp Maggiore 41: 431437, 1953.. Hertle L, and Androulakakis P: Keratinizing desquama-

    e squamous metaplasia of the upper urinary tractleukopla-cholesteatoma. J Urol 127: 631635, 1982.. Weitzner S: Cholesteatoma of the calix. J Urol 108: 365, 1972.. Willis JS, Pollack H, and Curtis JA: Cholesteatoma of the

    per urinary tract. AJR Am J Roentgenol 136: 941944,1.. Taguchi Y, Kotha V, Tomka B, et al: Conservinghrons in cholesteatoma. J Urol 123: 258260, 1980.. Neerhut G, Politis G, Alpert L, et al: Cholesteatoma ofrenal pelvis: endoscopic management. J Urol 139: 10324, 1988.. Gale GL, and Kerr WK: Cholesteatoma of the urinary

    ct. J Urol 104(1): 7172, 1970.. Lupovitch A, Domzalski H, and Tippins R: Cholestea-a of the renal pelvisa case with long term follow up.

    rol 140: 360361, 1988.

    FIGURE 3. Percutaneous approach.UROLOGY 64 (4), 2004

    LATE RECURRENCE OF RENAL CHOLESTEATOMA AFTER 15 YEARSCASE REPORTCOMMENTREFERENCES

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