RENAL COLLECTING DUCT CARCINOMA AND CONCOMITANT BLADDER UROTHELIAL CARCINOMA: A CASE REPORT

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1 REAL COLLECTIG DUCT CARCIOMA AD COCOMITAT BLADDER UROTHELIAL CARCIOMA: A CASE REPORT Hsi-Lin Hsiao, 1 Hsin-Chih Yeh, 1 Tu-Hao Chang, 1 Hung-Lung Ke, 1 Han-Ching Lin, 1 Wen-Jeng Wu, 1,2 Chun-Hsiung Huang, 1,2 and Yung-Chin Lee 1 1 Department of Urology, Kaohsiung Medical University Hospital, Kaohsiung Medical University, and 2 Department of Urology, Faculty of Medicine, College of Medicine, Kaohsiung Medical University, Kaohsiung, Taiwan. The simultaneous occurrence of renal collecting duct (Bellini duct) carcinoma and bladder urothelial carcinoma is very rare. A 68-year-old female patient with bladder urothelial carcinoma first received transurethral resection of the tumor. Left side nephrouretectomy and transurethral resection of the intramural ureter were performed 8 months later under the diagnosis of concomitant renal pelvic urothelial carcinoma. However, the final pathologic examination revealed collecting duct carcinoma. The patient received systemic chemotherapy after surgery, but distant metastasis was noted 6 months later. Here, we report a rare case of combined renal collecting duct carcinoma and bladder urothelial carcinoma confirmed by pathologic examination and immunohistochemical staining. Key Words: collecting duct carcinoma, urothelial carcinoma (Kaohsiung J Med Sci 2008;24:157 62) Collecting duct carcinoma (CDC) or Bellini duct carcinoma is a very rare and aggressive renal neoplasm that arises from the principle cells lining the distal collecting duct (Bellini duct) epithelium and the distal renal tubules, unlike the much more common variants of renal cell carcinoma (RCC), which arise from the convoluted tubules [1,2]. CDC accounts for approximately % of all RCC and tends to occur in younger (median age at diagnosis of 43 years), male (male-to-female ratio of 2:1) and white patients [3 7]. The term Bellini duct carcinoma was first coined by Cromie et al [8]. Of all Received: Apr 7, 2007 Accepted: May 17, 2007 Address correspondence and reprint requests to: Dr Yung-Chin Lee, Department of Urology, Kaohsiung Medical Hospital, 100 Tzyou 1 st Road, Kaohsiung 807, Taiwan. tracyhsiao9@yahoo.com.tw renal epithelial neoplasms, CDC is the most aggressive. Due to the rarity of this tumor and the lack of clinical awareness, no diagnostic protocol has been established. Concurrent RCC and urothelial carcinoma (UC) in one person is rare and has seldom been reported [9 13]. Here, we report a very rare, newly diagnosed case with combined bladder UC and renal CDC. CASE PRESETATIO A 68-year-old female patient was admitted to our hospital in August 2005 because of intermittent painless hematuria and left flank soreness. Abdominal ultrasonography revealed a left renal mass and hydronephrosis. Cystoscopic examination revealed a bladder tumor and left side retrograde pyelography revealed a filling defect in the left renal pelvis with hydronephrosis and hydroureter (Figure 1). Computed tomography Kaohsiung J Med Sci March 2008 Vol 24 o Elsevier. All rights reserved.

2 H.L. Hsiao, H.C. Yeh, T.H. Chang, et al (CT) revealed thickening of the posterior aspect of the bladder wall with an uneven surface and heterogeneous enhanced soft tissue in the left renal pelvis with renal parenchymal invasion (Figures 2 and 3). The renal contour remained normal in the image study. Transurethral resection of the bladder tumor was performed as well as a left side ureteroscopic biopsy with washing cytology. The results of pathologic examination of the bladder tumor revealed high-grade noninvasive papillary UC, but the renal pelvis showed chronic inflammation. However, cytologic examination of the washing cytology showed a papillary urothelial neoplasm of low malignant potential. Left nephrouretectomy with transurethral resection of the intramural ureter and lymph node dissection were performed in April Grossly, the tumor was diffusely distributed in the upper pole of the renal parenchyma with a poorly defined margin and whitish color (Figure 4). Microscopically, it showed multifocal tumor growth with a tubular or tubulopapillary and solid pattern in which irregular angulated glands infiltrated the renal parenchyma (Figure 5). The tumor cells were hyperchromatic, pleomorphic and had distinct nucleoli. Intratubular epithelial atypia adjacent to the tumor was also seen. Immunohistochemical studies demonstrated positive staining for cytokeratin (CK), Figure 1. Left side retrograde pyelography shows a left renal pelvis filling defect with hydroureter. Figure 2. Computed tomography shows the posterior aspect of bladder wall thickening with an uneven surface. A B Figure 3. Computed tomography shows heterogeneously enhanced soft tissue with renal parenchymal invasion in the left side renal pelvis. 158 Kaohsiung J Med Sci March 2008 Vol 24 o 3

3 Renal collecting duct carcinoma Figure 4. The tumor, whitish in color with a poorly defined margin, is diffusely distributed in the renal parenchyma, mainly in the upper pole. Figure 5. There is multifoci tumor growth with a tubular, tubulopapillary or solid pattern, in which irregularly angulated glands infiltrate the renal parenchyma (hematoxylin & eosin, 10 ). A B C D Figure 6. (A) Positive cytokeratin staining. (B) Positive staining for high-molecular-weight cytokeratin. (C) Focal positive staining for CD10. (D) egative staining for vimentin. high-molecular-weight CK (HMWCK) and CD10 (focal positive), and negative staining for vimentin (Figure 6). The results of histologic and immunohistochemical studies were consistent with CDC. The perirenal fat and lymph nodes showed tumor invasion. Kaohsiung J Med Sci March 2008 Vol 24 o 3 The patient was discharged on the seventh postoperative day and started receiving systemic chemotherapy. The chemotherapy regimen that we chose was active for the UC. Unfortunately, distant metastasis was noted 6 months after surgery. 159

4 H.L. Hsiao, H.C. Yeh, T.H. Chang, et al DISCUSSIO The idea that the collecting duct epithelium may be a source of tumor development was first raised by Mancilla-Jimenez et al in 1976 based on their finding of atypical and hyperplastic changes in the adjacent duct [14]. Patients with CDC are usually found to have a family history of associated malignancies, including colon, pancreas, lung, ovary and uterus cancers [15]. Hematuria, flank pain and a palpable mass are the most common presenting features of CDC. Other symptoms, such as fever, anorexia and body weight loss, are also common, but no particular paraneoplastic syndrome has been reported. CT scans generally reveal that affected kidneys maintain a normal contour with minimal contrast enhancement, and sometimes renal masses protruding into the central sinuses may also be noted [16]. Magnetic resonance imaging reveals heterogeneous low intensity masses [17]. Renal angiography usually reveals hypovascular or avascular tumors [18,19]. A definitive diagnosis can only be made by a detailed histopathologic examination [2]. Pathologically, the tumor is an adenocarcinoma and consists of two components. The main tumor is papillary with a fibrovascular core, and the invasive part is composed of glandular elements associated with a marked desmoplastic reaction. Cytologically, CDC shows malignant round-to-oval tumor cells with a modest eosinophilic cytoplasm and a predominantly papillary growth pattern. The tumor cells are columnar and show variable degrees of pleomorphism. The histologic diagnosis of CDC can be confirmed by positive immunohistochemical staining with a collecting duct marker, such as UEA-1 (Ulex europaeus agglutinin-1) or HMWCK, and negative staining for vimentin [20]. CDC must be differentiated from some centrally located renal lesion with infiltrative features, including invasive transitional cell or squamous cell carcinoma, lymphoma, renal metastasis, invasive RCC involving a column of Bertin, mesoblastic nephroma, renal medullary carcinoma and bacterial pyelonephritis [21]. The prognosis of CDC is generally poor because of its aggressive behavior and the advanced stage at the time of diagnosis. The standard treatment for CDC of the kidney is radical nephrectomy because no standard chemotherapy or immunotherapy regimen has been effective in the management of CDC. Because of the close relationship between CDC and UC (a common embryologic origin that arises from 160 the mesonephrons), chemotherapy may be considered in the treatment of CDC [22]. Bladder UC is not rare in adults, but only a few cases of CDC have been reported. The case of bladder UC and concomitant renal pelvis CDC that we report is very rare, even though the affected tissues have a common embryologic origin. Because of the poor prognosis of the disease, distant metastasis was noted 6 months after the operation. Typically, CDC has an aggressive course and many patients present with metastasis soon after diagnosis. In this paper, we report a newly diagnosed bladder UC simultaneously with a renal CDC treated with left nephrouretectomy plus transurethral resection of the intramural ureter and transurethral resection of the bladder tumor. Considering the aggressive character of this tumor, systemic chemotherapy was arranged for the patient after surgery, but unfortunately, distant metastasis occurred 6 months later. Due to the rarity and poor prognosis of this kind of tumor, we need more experience to be able to offer more effective management and detect such tumors at an earlier stage. REFERECES 1. Auguet T, Molina JC, Lorenzo A, et al. Synchronous renal cell carcinoma and Bellini duct carcinoma: a case report on a rare coincidence. World J Urol 2000;18: Dobronski P, Czaplicki M, Kozminska E, et al. Collecting (Bellini) duct carcinoma of the kidney clinical, radiologic and immunohistochemical findings. Int Urol ephrol 1999;31: Rumplet HJ, Storkel S, Moll R, et al. Bellini duct carcinoma: further evidence for this rare variant of renal cell carcinoma. Histopathology 1991;18: De Diego RE, Pascula SC, Gutierrez Banos JL, et al. Bellini s carcinoma. Our experience. Arch Esp Urol 2000; 53: Kasper HU, Buhtz P, Kruger G, et al. Bellini duct carcinoma of the kidney a case report. Gen Diagn Pathol 1997;143: Montserrat OV, Lopez BE, Perez BF, et al. Carcinoma of Bellini s ducts: apropos of a case. Arch Esp Urol 1997; 50: Dimopoulos MA, Logothetis CJ, Markowitz A, et al. Collecting duct carcinoma of the kidney. Br J Urol 1993; 71: Cromie WJ, Davis CJ, DeTure FA. Atypical carcinoma of kidney: possibly originating from collecting duct epithelium. Urology 1979;13: Kaohsiung J Med Sci March 2008 Vol 24 o 3

5 Renal collecting duct carcinoma 9. Yuichi A, Yoko K, Chiaki I, et al. A case of simultaneous contralateral renal cell carcinoma and ureteral transitional cell carcinoma. Urol Int 1991;46: Lee JW, Kim MJ, Song JH, et al. Ipsilateral synchronous renal cell carcinoma and transitional cell carcinoma. J Korean Med Sci 1994;9: Hong SK, Jeong SJ, Lee SE. A case of renal transitional cell carcinoma associated with synchronous contralateral renal cell carcinoma. J Korean Med Sci 2001;16: Straub B, Muller M, Schrader M, et al. Concomitant spread of a renal cell carcinoma beyond the kidney and a transitional cell carcinoma beyond the bladder. Int Urol ephrol 2000;32: Wegner HE, Bornhoft G, Dieckmann KP. Renal cell cancer and concomitant transitional cell cancer of the renal pelvis and ureter in the same kidney report of 4 cases and review of the literature. Urol Int 1993;51: Mancilla-Jimenez R, Stanley RJ, Blath RA. Papillary renal cell carcinoma: a clinical, radiologic and pathologic study of 34 cases. Cancer 1976;38: Kuroda, Toi M, Hirol M, et al. Review of collecting duct carcinoma with focus on clinical and pathobiological aspects. Histol Histopathol 2002;17: Fukuya T, Honda H, Goto K, et al. Computed tomographic findings of Bellini duct carcinoma of the kidney. J Comput Assist Tomogr 1996;20: Oida T, Takenawa J, Ogura K, et al. Collecting duct carcinoma (Bellini duct carcinoma) of the kidney with tumor extension into the inferior vena cava. Hinyokika Kiyo 1999;45: atsume O, Ozono S, Futami T, et al. Bellini duct carcinoma: a case report. Jpn J Clin Oncol 1997;27: Chatelain D, de Pinieux G, Slama J, et al. Renal medullary carcinoma, a new clinicopathological entity. Immunohistochemical, ultrastructural, flow cytometric and cytogenetic study of a case. Ann Pathol 1999;19: Kutta A, Schoenfeld B, Martin W, et al. Multifocal renal cell carcinoma of collecting duct origin. Scand J Urol ephrol 1993;27: Pickhardt PJ, Siegel CL, McLarney JK. Collecting duct carcinoma of the kidney: are imaging findings suggestive of the diagnosis? AJR Am J Roentgenol 2001;176: Srigley JR, Moch H. Carcinoma of the collecting ducts of Bellini. In: Eble J, Sauter G, Epstein JI, et al, eds. WHO Classification of Tumors of the Urinary System and Male Genital Organs. Lyon: IARC Press, 2004:33 4. Kaohsiung J Med Sci March 2008 Vol 24 o 3 161

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