Low-grade Collecting Duct Carcinoma Research Articles

Expression of Aldo-keto-reductase 1B1 (AKR1B1) in tubulocytic carcinoma of kidney.

507 Background: Tubulocystic carcinoma of the kidney (TubCC) is a new entity of tumor not listed in the 2004 WHO classification. The tumor comprised tubules forming duct-like structures exhibiting hobnail change, and resembled collecting duct carcinoma (CDC) by immunostaining; thus, this tumor was named low grade CDC. Since then, similar tumors have been reportedly called tubulocystic carcinoma (TubCC)We explored the possibility whether Aldo-keto-reductase-1B1 (AKR1B1), osmoregulatory protein and abundant in collecting duct, can be used as a marker for the pathologic diagnosis of TubCC. Methods: We prepared anti-AKR1B1 monoclonal antibodies, examined expression of AKR1B1 protein in the human normal kidney. Immunohistochemical expressions of AKR1B1 was examined on normal kidney, 4 cases of TubCC, 10 cases of papillary renal cell carcinoma (pRCC), and 10 cases of invasive urothelial carcinoma (iUC). Results: AKR1B1 expression distributed preferentially in the renal medulla and not in the cortex, reflected by its high concentration in medulla (2.12 ±1.35 μg/mg) compared with cortex (0.13 ± 0.03 μg/mg). Immunohistochemically, AKR1B1 was strongly positive in 3 of 4 TubCC cases. AKR1B1 was negative in cases of either iUC or pRCC. Conclusions: Although the present study was small and preliminary, AKR1B1 may be a more specific and potentially useful marker for TubCC. [Table: see text]

Tubulocystic Carcinoma of the Kidney

Tubulocystic carcinoma (TCC) of the kidney is a unique, rare, and recently recognized neoplasm. Although originally considered a low-grade collecting duct carcinoma, TCC is now considered to be a distinct entity. TCC should be considered in the differential diagnosis of cystic renal neoplasms. We report a case of TCC arising in the left kidney.

Gains of chromosomes 7 and 17 in tubulocystic carcinoma of kidney: two cases with fluorescence in situ hybridisation analysis

Tubulocystic carcinoma (TCC) is a very rare renal tumour with unique gross and microscopic features, alternatively considered as low-grade collecting duct carcinoma. Recent studies favoured distinction of TCC from collecting...

Tubulocystic carcinoma of the kidney

Examples of tubulocystic carcinoma of the kidney were introduced in an abstract presented at the Annual Meeting of the United States and Canada Academy of Pathology1 and also in the Armed Forces Institute of Pathology Atlas of Tumor Pathology, Series III, in 1994.2 However, the morphological features of this rare distinctive renal tumor were described in 1955 byMasson, who designated the lesion a “Bellinian epithelioma” because he regarded it as a neoplasm originating in the collecting ducts of Bellini. The first detailed report of the clinical and pathological details of tubulocystic carcinoma were published in 1997.3 The authors described the neoplasm as a “low-grade mucinous tubulocystic renal carcinoma of possible collecting duct origin”. The cases were initially considered to be derived from the collecting duct; however, since those reported tumors were of a low-grade malignancy, the behavior of which significantly differed from the classical highly aggressive collecting duct carcinomas, the term “low-grade collecting duct carcinoma” was coined for this peculiar renal tumor. Thereafter, with more cases showing similar clinical and pathological features, the name of the neoplasm was designated “tubulocystic carcinoma” to denote the specific morphology.4e8 To date, more than 50 cases have been reported. A review of the literature showed a male predominance (of around 7:1) with patient ages ranging from 34 years to 74 years. Tumor sizes range from 0.3 cm to 17 cm, but most are small and discovered incidentally. Tumors tend to present in a low stage (pT1a) and behave indolently. However, sarcomatoid change,9 and metastases to lymph nodes, lungs, and brain were reported.7

腎Tubulocystic carcinomaの1例

A 55-year-old man who presented himself with gross hematuria and right back pain was found to have a right renal mass with evidence of metastasis to the lymph nodes, bone and lung (cT1bN1M1). He underwent a transperitoneal right nephrectomy. Tumor expressed markers of CD10, P504S and CK19 immunohistochemically, so histopathological examination revealed tubulocystic carcinoma of the right kidney (pT3a). After the patient received sunitinib therapy, computed tomography revealed reduction in the size of the metastatic lung nodule and lymph nodes, indicating a partial response. He is alive without disease progression at 12 months after nephrectomy. Tubulocystic carcinoma has been referred to by Amin et al as low-grade collecting duct carcinoma and is not yet included in the World Health Organization (WHO) 2004 classification of renal tumors. The cells lining the tumor range from cuboidal to columnar and have large nuclei with low-grade changes and abundant eosinophilic or amphophilic cytoplasm. Hobnail cells are commonly seen. Immunohistochemically, tubulocystic carcinomas are strongly positive for markers of the proximal nephron (CD10, P504S) and the distal nephron (parvalbumin, CK19). Despite a low nuclear grade, tubulocystic carcinomas occasionally show progressive behavior clinically. Although there is no established salvage therapy, sunitinib was found to be effective for this patient.

Tubulocystic Carcinoma of the Kidney: A Case Report of Natural History and Long-Term Follow-Up

Tubulocystic carcinoma (TC) is a rare primary renal tumor that has been recently described in the pathology literature. Formerly termed low-grade collecting duct carcinoma, further molecular analysis has shown TC to be a distinct entity that is separate from the more aggressive collecting duct carcinoma. Previous series have described the microscopic and immunohistochemical features of this tumor. We describe the natural history of this tumor in a patient who was followed with active surveillance for several years and then underwent partial nephrectomy. Long-term follow-up has shown no evidence of disease. A review of the pertinent literature is performed.

Comparison of Gene Expression Profiles in Tubulocystic Carcinoma and Collecting Duct Carcinoma of the Kidney

The relationship between tubulocystic carcinoma and collecting duct carcinoma of the kidney remains controversial. Some experts are of the opinion that the tumors are related, considering tubulocystic carcinoma to be synonymous with low-grade collecting duct carcinoma. However, others maintain that the 2 are distinct, unrelated entities on the basis of morphologic features and clinical outcome. To explore the relationship between tubulocystic carcinoma and collecting duct carcinoma, we compared the expression of several gene products at the mRNA level in cohorts of each tumor subtype. Seven cases of tubulocystic carcinoma and 8 cases of collecting duct carcinoma were identified. Total RNA was isolated from formalin-fixed paraffin-embedded tissue from each case. Relative expression levels of vimentin, alpha methylacyl CoA racemase, E-cadherin, p53, CD10 antigen, parvalbumin, cytokeratin 7, and cytokeratin 19 were assessed by quantitative reverse transcription polymerase chain reaction. Tubulocystic carcinoma was characterized by relative overexpression of vimentin, p53, and alpha methylacyl CoA racemase, compared with collecting duct carcinoma (P<0.05 for each gene, t test). In general, tubulocystic carcinoma expressed higher levels of E-cadherin and CD10, whereas collecting duct carcinoma expressed higher levels of cytokeratin 19; however, these trends did not reach statistical significance in this study cohort. Tubulocystic carcinoma and collecting duct carcinoma did not express cytokeratin 7 differentially. Case-to-case variability of gene expression limited the effectiveness of any one marker to distinguish the tumor types. Our study demonstrates that tubulocystic carcinoma and collecting duct carcinoma have different expression profiles of selected genes, including vimentin, p53, and alpha methylacyl CoA racemase. Further analysis of additional cases, using quantitative reverse transcription polymerase chain reaction and immunohistochemistry, will be useful to test the reproducibility of these findings. In addition, larger studies may establish statistical differences in expression of other genes analyzed in this study. Overall, these findings support the view that tubulocystic carcinoma and collecting duct carcinoma should be considered as 2 distinct entities at the molecular level.

A Case of Tubulocystic Carcinoma Simultaneously Occurring with Clear Cell Type Renal Cell Carcinoma and Micropapillary Urothelial Carcinoma of Bladder

The coexistence of multiple and synchronous primary neoplasms in the genitourinary system has rarely been described in the literature. To our knowledge, this is the first report of a very rare tumor, a "tubulocystic carcinoma," simultaneously occurring with micropapillary urothelial carcinoma in the bladder and clear cell type renal cell carcinoma in the same kidney. Tubulocystic carcinoma is a recently entitled tumor. It had been thought to arise from the distal collecting duct for a period of time and was therefore named as a low-grade collecting duct carcinoma. Microscopically, the tumor is defined as being tubulocystic or having tubulopapillary architecture and a low nuclear grade with hobnail morphology. Its progression and treatment protocol are uncertain.

Tubulocystic Carcinoma of the Kidney

A distinctive tumor described under the terms Bellini duct carcinoma and low-grade collecting duct carcinoma has been referred to by us and others as tubulocystic carcinoma. This renal cell carcinoma subtype is not recognized in the World Health Organization 2004 classification. Herein, we present a detailed study of 31 cases to further characterize this rare subtype of renal cell carcinoma. The tumor occurred in adults (mean age, 54 years) with a strong male predominance (7:1). Grossly, the tumors ranged from 0.7 to 17 cm, and exhibited a spongy or "bubble wrap" appearance reflecting the microscopic presence of variably sized cystically dilated tubules lined by a single layer of epithelium. The lining varied with a cuboidal, flat, and hobnail cell appearance, and the neoplastic cells had abundant eosinophilic cytoplasm and enlarged nuclei with prominent nucleoli. The cysts were closely spaced with an intervening variably fibrotic stroma. Immunohistochemistry and ultrastructural examination showed features of proximal convoluted tubules (Pax 2 immunoreactivity and short microvilli with brush border organization) and distal nephron (kidney-specific cadherin immunoreactivity and cytoplasmic interdigitation). Gene expression profiling showed that tubulocystic carcinoma displayed a unique molecular signature. Twenty-four tumors were stage pT1, 4 stage pT2, and 3 stage pT3. Disease progression (median follow-up of 56 months) occurred in 3 patients; 1 with local recurrence, and 2 with distant metastasis to bone and liver. In light of the distinctive clinicopathologic features and a low but definite metastatic potential, this unique subtype of renal cell carcinoma deserves formal recognition in the contemporary classification of renal neoplasms.

Tubulocystic Carcinoma of the Kidney

The nature of tubulocystic carcinoma, a rare renal tumor composed of tubular and cystic structures, is poorly understood. It has been suggested that it may represent a low-grade collecting duct carcinoma of the kidney despite the lack of sufficient molecular and pathologic evidence. The aim of this study was to examine the clinical and pathologic features of 13 cases of tubulocystic carcinoma of the kidney. Furthermore, using gene expression microarray analysis, we defined the molecular signature of this tumor by comparing it with other renal tumors in our previously established molecular profile database. Histologically, all 13 tumors were composed of closely packed tubules and cysts of varying sizes separated by fibrovascular septa. The epithelial lining cells of the tubules and cysts in this tumor were characterized by abundant eosinophilic cytoplasm with prominent nucleoli often showing a hobnail appearance. Clinically, one of the 13 cases showed metastasis to the pelvic lymph nodes. Five of the 13 cases coexisted with papillary renal cell carcinoma (RCC) (n=3) or papillary adenoma (n=2). In addition, the molecular profile of tubulocystic carcinoma was similar but not identical to those of papillary RCC by clustering analysis. Through comparative genomic microarray analysis, tubulocystic carcinoma showed gains of chromosome 17, but not chromosome 7, whereas most papillary RCCs showed chromosomal gains in both 7 and 17 (trisomies). Therefore, based on its unique pathologic features and molecular signature as well as its biologic behavior to develop metastasis either by itself or in association with papillary RCC, tubulocystic carcinoma of the kidney should be recognized as a distinct subtype of RCC and be distinguished from other malignant and benign cystic lesions of the kidney.

Carcinoma renal de los conductos colectores de bajo grado en un paciente en tratamiento con hemodiálisis

Patient treated by dialysis for end-stage renal disease are at increased risk of many cancers, especially those of kidney and urinary tract. A 56 years old man who undergoing chronic dialysis, presented in ultrasonography a 2 cm complex cystic mass in the left kidney what was confirmed by computed tomography and magnetic resonanceA laparoscopic left nephrectomy was performed. Histological examination showed a low grade collecting duct carcinoma of the kidney. An infrequent variant of kidney tumor

Pseudotumor fibroso testicular: Aportación de un caso

We report a case of fibrous pseudotumor of the epididymis as a even more rare location of nodular, fibrous and ossification diffuse proliferation, it may be difficult to distinguish from solid tumors. We also reviewed published report up to date and the differential diagnosisA laparoscopic left nephrectomy was performed. Histological examination showed a low grade collecting duct carcinoma of the kidney. An infrequent variant of kidney tumor

Distribution of Cytokeratins and Vimentin in Adult Renal Neoplasms and Normal Renal Tissue

Adult renal epithelial neoplasms (RENs) comprise several distinct clinicopathologic entities with potential prognostic and therapeutic differences. Individual cases can show overlapping morphologic features, necessitating the use of ancillary methods. The purpose of this study was to determine the diagnostic utility of cytokeratin (CK) subtype expression pattern in a wide range of adult RENs. RENs (including clear cell [conventional] renal cell carcinoma (RCC), papillary RCC, chromophobe RCC, renal oncocytoma, collecting duct carcinoma (CDC), renal medullary carcinoma (RMC), urothelial carcinoma, metanephric adenoma (MA), tubulocystic carcinoma (TC) (also known as low-grade collecting duct carcinoma), and mucinous tubular and spindle cell carcinoma) were immunostained for CK subtypes (CK5/CK6, 7, 8, 13, 14, 17, 18, 19, 20), high molecular weight CKs 1, 5, 10, 14 (HMWCK), and vimentin (Vim). The expression pattern of normal kidney was also examined and correlated with RENs. Although there is some overlap, subtypes of RENs show distinctive CK expression profiles that may be useful in several differential diagnostic settings. Clear cell RCCs typically showed a restricted expression pattern of CK8, CK18 and Vim. Papillary RCCs typically expressed CK7, CK8, CK18, CK19, and Vim and could be distinguished from MA (CK7-). Chromophobe RCCs were typically CK7+, CK8+, CK18+, and Vim-, and could be distinguished from oncocytomas (typically CK7-). In oncocytomas, nonspecific staining of unblocked endogenous biotin is a potentially significant diagnostic pitfall. CDC, RMC, and TC demonstrated similar CK expression profiles (with the exception of HMWCK expression limited to CDC), supporting a close relationship between these entities. A panel of CK5/CK6, CK17, and Vim may be helpful in distinguishing CDC (typically CK5/CK6-, CK17-, Vim+) and urothelial carcinoma (typically CK5/CK6+, CK17+, Vim-). In conclusion, CK expression patterns may be helpful in several differential diagnostic situations when dealing with adult RENs.

Review of mucinous tubular and spindle-cell carcinoma of the kidney with a focus on clinical and pathobiological aspects.

Recently, the characterization of mucinous tubular and spindle-cell carcinoma (MTSCC) has been established. MTSCC predominantly occurs in females. This tumor is histologically characterized by eosinophilic cytoplasm, elongated and anastomosing tubules, myxomatous stroma and low-grade nuclear cytology. Proliferation of spindle cells or foci of clear cells are also observed. Histochemically, the myxomatous stroma exhibits a positive reaction for alcian blue and colloidal iron stainings. Ultrastructurally, short microvilli are focally observed and junctional complexes are present. Recently, multiple losses of chromosomes 1, 4, 6, 8, 9, 13, 14, 15 and 22 in MTSCC have been elucidated by using comparative genomic hybridization. The prognosis of MTSCC is generally favorable, but some cases may show local recurrence or metastasis. Some cases with MTSCC seem to show overlapping histology with low-grade collecting-duct carcinoma. Therefore, further investigation will be needed to elucidate pathobiological characteristics of MTSCC.

Carcinoma renal de los conductos colectores de bajo grado. presentación de un caso y revisión de la literatura

Low grade collecting duct carcinoma of the kidney. presentation of one case and literature revisionCollecting duct renal cell carcinoma is an uncommon variant of renal carcinoma. Typically its behaviour is more aggressive than other forms of renal carcinoma and usually it is diagnosed at advanced stages. A 57-year-old man visited our hospital by right lumbar pain. Abdominal CT showed a enhanced mass on the right kidney. A right partial nephrectomy was done. Histological examination of the surgical specimen showed a low grade collecting duct carcinoma of the kidney. This is a new entity, with unknown behaviour but seems to be less aggressive than classical collecting duct carcinoma of the kidney. The clinical, radiological, pathological and immunohistochemical characteristics of this tumor are reported, and the literature is reviewed.

Collecting duct carcinoma of the kidney

The collecting duct carcinoma that we reported 1 showed tubulopapillary growth pattern and low-grade nuclear atypia (Fuhrman’s grade 2), 2 clearly bordered by fibrous capsule. It was unusual in typical collecting duct carcinoma. Even though it was not mentioned in our paper, the immunohistochemical study showed positive reaction against UEA-1, CAM 5.2, BerEP4 and Progen19. Alcian blue was positive in the aliquot area. More importantly, this small neoplasm (1.5 cm in diameter) existed clearly in the renal medulla. We believed that the tumor color was influenced by the preoperative needle biopsy; therefore, we synthetically diagnosed it as collecting duct carcinoma. In Japan, regular health check-ups among middleaged and elderly people are popular. Therefore, the detection rate of incidental carcinoma may be higher than that in other countries. Subsequently, there is the possibility that the tumors are diagnosed at an early stage, and we may find the clinical and pathological features such as those in our previous paper. Srigley and Eble 3 have proposed major and minor diagnostic criteria for collecting duct carcinoma, and have presented a scheme of the spectrum of collecting duct carcinoma. Papillary carcinomas also show positive reaction for distal nephron markers, which may make it difficult to distinguish between low-grade collecting duct carcinoma and papillary carcinoma. Additional pathologic series should help to elucidate the morphologic and biological spectrum of collecting duct neoplasia. Furthermore, cytogenetic and molecular genetic studies 4–7 may help sort out the morphologic diversity of putative collecting duct carcinomas, very much like the way that molecular studies have assisted in the classification of other renal epithelial neoplasms including clear cell, papillary and chromophobe renal cell carcinomas.

Low-grade Collecting Duct Carcinoma Of The Kidney: A Potential Mimic Of Conventional Clear Cell Carcinoma

Low-grade Collecting Duct Carcinoma Of The Kidney: A Potential Mimic Of Conventional Clear Cell Carcinoma

Low-grade Collecting Duct Carcinoma Of The Kidney: A Potential Mimic Of Conventional Clear Cell Carcinoma

Collecting duct carcinoma of the kidney is a well-known, but relatively uncommon tumor characterized by a high cytologic grade, desmoplastic stroma and a poor clinical outcome. Since 1979, however, there have been several reports of low-grade renal carcinoma of putative collecting duct origin. These low-grade tumors were extensively papillary, tubulopapillary or tubulocystic, with a low nuclear grade (usually Furhman grade 1 or 2). Most have conferred a benign follow-up course, even when treated only by partial nephrectomy. The previous reports of such tumors have been variably characterized immunohistochemically and ultrastructurally. We report the cases of a low-grade renal carcinoma of collecting duct origin, arising in the right renal lower pole of a 47-year-old-male with a history of gout and nephrolithiasis. Microscopically, the tumor showed a tubular and tubulocystic pattern and was composed almost exclusively of clear cells, closely mimicking conventional (clear cell) renal cell carcinoma. Tumor cells stained positively for PAS, showed droplet-like staining with Hale\'s colloidal iron, were variably alcian blue positive and were mucicarmine negative. They were reactive with immunohistochemical antibodies to peanut agglutinin (PNA), Ulex europaeus-1 agglutinin (UEA-1), pan-cytokeratin, CAM 5.2, cytokeratin 7, epithelial membrane antigen (EMA) and vimentin. Electron microscopy showed microvillous-lined tubules, desmosomes, and tonofilaments, with a paucity of mitochondria. The patient was treated by partial nephrectomy and has been without evidence of disease for a follow-up period of 10 months.

Low-grade collecting duct carcinoma of the kidney: report of 13 cases of low-grade mucinous tubulocystic renal carcinoma of possible collecting duct origin

To assess the nature of a series of unusual low-grade mucin-producing tubulocystic renal cancers diagnosed at the Mayo Clinic since 1985. We reviewed the clinical, radiologic, and pathologic features of 13 unusual low-grade renal carcinomas with features most suggestive of collecting duct origin. In 8 cases, the tumor was discovered incidentally. Presenting symptoms in the other 5 patients were similar to those of typical renal carcinoma. Imaging studies and angiography disclosed solid, cystic, or complex masses that were relatively avascular. Pathologic assessment revealed good circumscription, minimal hemorrhage and/or necrosis, minimal tendency to extend beyond the kidney, tubulocystic architecture, a fibrotic interface with adjacent normal renal parenchyma, low nuclear grade, and minimal mitotic activity. Mucin production was noted in all evaluable cases. All tumors expressed keratins AE1/AE3 and/or Cam 5.2. The tumors showed immunoreactivity to antibodies directed against keratin 34 beta E12 (8 of 13 cases), and Ulex Europeus antigen 1 (6 of 10 cases). Follow-up ranged from 12 to 114 months (mean 62). No metastases occurred in 12 patients. One patient died of metastatic carcinoma morphologically identical to the primary renal neoplasm 46 months after his tumor had been misinterpreted as a benign condition. We believe the tumors described in this article may be of collecting duct origin, representing the low-grade end of a spectrum of cancers arising in collecting duct epithelium.

Nephrogenic Adenofibroma

Nephrogenic adenofibroma is a novel kidney tumor of young people (mean age of presentation, 13 years), who present with polycythemia, hypertension, or hematuria, which resolve following nephrectomy. The typical nephrectomy specimen contains a solitary, nonencapsulated, vaguely circumscribed, irregularly shaped or spherical, firm mass with either tan, gray-white, or pale yellow coloration. Cysts are sometimes present within the tumor. The histologic appearance is distinctive and characterized by a marked proliferation of spindled mesenchymal cells resembling the classical type of congenital mesoblastic nephroma, encasing discrete nodules of embryonal epithelium similar to the hyperplastic nephrogenic rests (nephroblastomatosis) usually associated with Wilms' tumor. The mesenchymal component consists of a fascicular proliferation of tightly interlaced, uniform, benign-appearing spindled cells that immunostatin for vimentin and fibronectin, but not desmin or actin. The epithelial component consists of discrete islands of blastemal cells that are partially or fully differentiated toward tubular, tubulopapillary, or papillary structures. Psammoma bodies are plentiful. Embryonal epithelium immunostains for cytokeratin but not epithelial membrane antigen. The overall histologic appearance of the mesenchymal and epithelial components is benign, and preliminary clinical data suggest that the tumor has a benevolent course. Two cases, however, contained small, well-circumscribed papillary lesions near the renal pelvis that resembled low-grade collecting duct carcinoma. The clinical implications of the latter finding are unclear.