|Year : 2010 | Volume
| Issue : 3 | Page : 339-341
Rare coexistence of keratinizing squamous cell carcinoma with xanthogranulomatous pyelonephritis in the same kidney: Report of two cases
Kavita Mardi, Vijay Kaushal, Vanita Sharma
Department of Pathology, Indira Gandhi Medical College, Shimla, India
|Date of Web Publication||29-Nov-2010|
12-A, Type V Quarters, GAD Colony, Kasumpti, Shimla, Himachal Pradesh
Squamous cell carcinoma of urinary tract is a rarely encountered tumor. It is more frequently reported in urinary bladder and male urethra than renal pelvis. Squamous cell carcinoma of renal pelvis is usually associated with nephrolithiasis. However, coexistence of keratinizing squamous cell carcinoma with xanthogranulomatous pyelonephritis is exceedingly rare with only one case on record so far. We report two such cases detected incidentally in patients who have undergone nephrectomy for hydronephrosis. The post operative histological evaluation revealed unsuspected squamous cell carcinoma of renal pelvis with concomitant xanthogranulomatous pyelonephritis.
Keywords: Kidney, renal pelvis, squamous cell carcinoma, xanthogranulomatous pyelonephritis
|How to cite this article:|
Mardi K, Kaushal V, Sharma V. Rare coexistence of keratinizing squamous cell carcinoma with xanthogranulomatous pyelonephritis in the same kidney: Report of two cases. J Can Res Ther 2010;6:339-41
|How to cite this URL:|
Mardi K, Kaushal V, Sharma V. Rare coexistence of keratinizing squamous cell carcinoma with xanthogranulomatous pyelonephritis in the same kidney: Report of two cases. J Can Res Ther [serial online] 2010 [cited 2013 May 23];6:339-41. Available from: http://www.cancerjournal.net/text.asp?2010/6/3/339/73351
| > Introduction|| |
Primary squamous cell carcinoma of renal pelvis and ureter constitute less than 1% of all urinary tract neoplasms.  Rare upper urothelial malignancies can be associated with renal stone disease or hydronephrosis ,, probably associated with a chronic inflammation or infection or with analgesic abuse.  In our cases, the malignancy was detected postoperatively during histopathological examination of nephrectomized specimen. We report two cases for rarity of their location (renal pelvis) and their coexistence with xanthogranulomatous pyelonephritis.
| > Case Reports|| |
A sixty-five-years-male presented with fever and pain in the left loin for two months. He had history of urolithiasis for a long time. Physical examination revealed tenderness on left costovertebral angle and palpable mass in left upper quadrant of abdomen. Blood glucose and renal function tests were normal. Ultrasonography showed evidence of hydronephrotic sac on left side with multiple stones. A left nephrectomy was performed. On gross examination of the nephrectomy specimen, cutsection of the enlarged kidney revealed lobules of yellow areas with focal grey white areas and dilated pelvicalyceal system containing multiple stones. [Figure 1]. Multiple sections from the various areas revealed dilated pelvicalyceal system lined by metaplastic squamous epithelium showing malignant transformation with nests of malignant squmous epithelium forming keratin pearls invading the underlying stroma and surrounding renal parenchyma that revealed features of xanthogranulomatous pyelonephritis.
|Figure 1: Cut section of the enlarged kidney showing predominantly yellowish areas with focal grey white and cystic areas|
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A sixty-years-old female presented with history of intermittent mild right flank pain. She had history of urolithiasis for a long time. Physical examination revealed slight tenderness on right costovertebral angle and palpable mass in right upper quadrant of abdomen. Hematological and biochemical parameters were within normal limits. A contrast enhanced computed tomographic (CECT) scan was done that suggested a right side hydronephrosis with multiple small calculi in dilated pelvicalyceal system. Patient underwent right nephrectomy.
On gross examination of the nephrectomy specimen, kidney was enlarged in size. Cut section showed dilated pelvicalyceal system containing multiple white colored stones. There was an irregular grey white growth arising from renal pelvis measuring 5 × 3 cm. Surrounding residual renal parenchyma showed yellowish areas. Microscopic examination revealed well differentiated squamous cell carcinoma with pearl formation invading adjacent renal parenchyma [Figure 2]. Areas of renal pelvis adjacent to growth showed squamous metaplasia. Perinephric fat was free from tumor invasion. Surrounding renal parenchyma showed features of xanthogranulomatous pyelonephritis with sheets of foamy macrophages, numerous foreign body giant cells, lymphocytes, plasma cells, occasional neutrophils, cholesterol clefts, and fibrosis in the interstitial tissue. [Figure 3] Diagnosis of well differentiated squamous cell carcinoma with xanthogranulomatous pyelonephritis was made.
|Figure 2: Photomicrograph showing areas of well differentiated squamous cell carcinoma (H and E, 40×)|
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|Figure 3: Photomicrograph revealing squamous cell carcinoma along with coexistent xanthogranulomatous pyenonephritis in the adjacent renal parenchyma (H and E, 20×)|
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| > Discussion|| |
Primary squamous cell carcinoma of renal pelvis and ureter constitute less than 1% of all urinary tract neoplasms.  These tumors are centered in renal pelvis and secondarily invade the renal sinus fat and renal parenchyma. In general, these tumors are highly aggressive and are at high stage when detected and can be expected to have poor clinical course. Lack of characteristic presentation like hematuria, pain and palpable mass causes delay in diagnosis. Most of the detected tumors are histologically high grade, 84% of the tumors are locally advanced or metastatic.  Women are affected more frequently than men predominantly in the age group of 50 to 70 years.
Tumors of the upper urinary tract and urinary bladder cancer share the same risk factors and can occur as a part of "field cancerization," which results from exposure of urothelium to carcinogens excreted by or activated in the urine. Hence, upper-urinary-tract urothelial tumors may be multifocal, and in 2-10% of cases, they are bilateral as well. Squamous cell carcinoma of the renal pelvis is often associated with phenacetin consumption, chronic renal calculi or infection.  Squamous metaplasia in the adjascent mucosa is seen in 17% to 33% of the patients.  Whether the occurrence of squamous metaplasia is due to the presence of the calculus that leads ultimately to the development of carcinoma or existence of squamous cell carcinoma causes the formation of calculus is not clear yet. This dilemma is particularly valid for the co-existence of the tumor and calculus at the renal pelvis. As renal squamous cell carcinoma is frequently associated with infected staghorn calculi existing for a long duration, it is recommended for the patients with renal stone disease who do not need surgery or patients with non-functioning kidney due to stone disease to be carefully examined with imaging modalities.
Workers in the chemical, petrochemical, aniline dye, and plastics industries, as well as those exposed to coal, coke, tar, and asphalt, are at increased risk for renal pelvis and ureteral tumors. Cigarette smoking appears to be the most significant acquired risk factor for upper-tract UC. It is suggested that 70% of upper-tract urothelial tumors in men and 40% in women can be attributable to smoking. Analgesic abuse is a risk factor; a combination of phenacetin use and papillary necrosis results in a 20-fold increase in risk for renal urothelial tumors. Chronic bacterial infection with urinary calculus and obstruction may predispose to development of urothelial cancer. SCC is the most common entity in these cases. Schistosomiasis also may predispose to SCC. The chemotherapy drugs like cyclophosphamide and ifosfamide are implicated in the development of renal pelvic tumors, particularly following drug-induced hemorrhagic cystitis.
Xanthogranulomatous pyelonephritis (XPN), a rare form of chonic pyelonephritis, is commonly associated with lithiasis and rarely leads to keratinizing squamous metaplasia.  Its manifestations closely mimic those of a renal neoplasm, leading to misdiagnosis of malignancy, often resulting in radical nephrectomy. As metaplasias are known to occur in association with calculi, its presence along with XPN is not surprising. However, only isolated incidence of such lesions is on record, where the metaplasia had already developed into squamous cell carcinoma. 
Establishing a diagnosis of squamous cell carcinoma can be arbitrary since some transitional cell carcinomas can have focal squamous differentiation. This subjective problem can be dealt by ensuring that histological material contains obvious and extensive squamous regions.
We have reported this case to highlight that a diagnosis of malignancy should be considered in a patient having long standing history of urolithiasis. As renal squamous cell carcinoma is frequently associated with infected staghorn calculi existing for a long duration, it is recommended for the patients with non-functioning kidney due to stone disease to be carefully examined with imaging modalities for early detection of the tumor, and warrants aggressive treatment with surgery followed by adjuvant aggressive combination chemotherapy that may provide a better outcome. If dissection appears difficult during nephrectomy for calculus with non-functional kidney, keeping a possibility of associated urothelial malignancy in mind, it is advisable to dissect in a plane outside Gerota's fascia as for radical nephrectomy. 
| > References|| |
|1.||Papadopoulos I, Wirth B, Jacobsen KW, Loch T, Wacker HH. Primary squamous cell carcinoma of ureter and adenocarcinoma of renal pelvis: 2 case reports. J Urol 1996;155:288-9. |
|2.||Sheaff M, Fociani P, Badenoch D, Baithun S. Verrucous carcinoma of the renal pelvis: Case presentation and review of the literature. Virchows Arch 1996;428:375-9. |
|3.||Fekak H, Rabii R, Moufid K, Joual A, Dahami Z, Mrini M. Unusual clinical presentations of tumours of the renal pelvis: Report of two cases. Prog Urol 2002;12:482-5. |
|4.||Ueno M, Jitsukawa S, Nakashima J, Ban S, Deguchi N. Primary transitional cell carcinoma in situ and renal calculus in the dilated pelvis. BJU Int 2002;89:324. |
|5.||Stewart JH, Hobbs JB, McCredie MR. Morphologic evidence that analgesic-induced kidney pathology contributes to the progression of tumours of the renal pelvis. Cancer 1999;86:1576. |
|6.||Nativ O, Reiman HM, Lieber MM. Treatment of primary squamous cell carcinoma of upper urinary tract. Cancer 1991;68:2575-8. |
|7.||Lee TY, Ko JF, Wan YL. Renal squamous cell carcinoma: CT Findings and clinical significance. Abdom Imaging 1998;23:203-8. |
|8.||Melamed MR, Reuter VE. Pathology and staging of urothelial tumours of kidney and ureter. Urol Clin North Am 1993;20:333-47. |
|9.||Dhingra KK, Sukard S, Shyama J. Rare coexistence of keratinizing squamous metaplasia with xanthogranulomatous pyelonephritis: Report of a case with the role of immunocytochemistry in the differential diagnosis. Acta Cytol 2007;51:92-4. |
|10.||Pitts JC, Peterson NE,Conley MC. Calcified functionless kidney in a 51 year old man. J Urol 1981;125:398-401. |
|11.||Wani B, Bole A, Yeola M. Rare upper urothelial malignancy in non-functioning nephrolithic kidney. Int J Urol 2009;6:1-6. |
[Figure 1], [Figure 2], [Figure 3]
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