個案報告：偽裝為膀胱腫瘤的黃色肉芽腫性膀胱炎

陳柏翰¹、鄭鼎耀¹、林祖鋒¹、許炯明¹

¹台灣基督長老教會馬偕醫療財團法人馬偕紀念醫院 泌尿科

A Case Report: Xanthogranulomatous Cystitis Masquerading Bladder Tumor

Bo-Han Chen¹, Ting-Yao Cheng¹, Tsu-Feng Lin¹, Jong-Ming Hsu¹

Department of Urology, MacKay Memorial Hospital, Taipei, Taiwan¹

Background

            Xanthogranulomatous change is a type of chronic granulomatous inflammation which commoly occurs in the gall bladder and kidneys. Xanthogranulomatous cystitis (XC) has been described as extremely rare. Fewer than 40 cases have been reported since first description in 1932. Patient often complain of unspecific lower urinary tract symptoms like frequency or urgency. Radiographically, it is difficult to distinguished from bladder urothelial cancer. Treatment includes antibiotic therapy and surgical resection. In this report, we present a case of XC masquerading bladder malignancy which was treated with transurethral resection.

Case presentation

               A 68-year-old woman presented with terminal hematuria with blood clot. She had medical history of hypertension, type 2 diabetes mellitus and end stage renal disease under hemodialysis for 1 year. No dysuria or abdominal discomfort was complained. Physical examination was unremarkable, and laboratory data reported anemia, elevated serum creatinine and elevated blood urea nitrogen. Urine analysis revealed 4 red blood cells and 12 white blood cells per high power field. Urine culture was negative. Renal echography revealed bilateral chronic parenchymal disease. She underwent cystoscope for painless gross hematuria. Three protruding tumors were found over the bladder neck, right lateral wall and left lateral wall of the bladder. (Figure 1) Enhanced computed tomography demonstrated three mass lesions compatible to the finding of cystoscope; no perivesical invasion or lymphadenopathy was noted. (Figure 2) A complete endoscopic resection with Thulium laser was performed under the impression of bladder neoplasm. Microscopic histopathology revealed chronic inflammatory xanthogranulomatous cystitis, including foamy macrophages that stained positive for CD68. (Figure 3) The patient made an uneventful recovery. Cystoscope was followed 3 months after the surgery and no recurrence was noted.

Discussion and conclusion

            Xanthogranulomatous processes are rare and chronic inflammatory condition, which has been described in many organ systems including gastrointestinal, urogenital, neurologic, and gynecologic. It is relatively common in kidney and gallbladder, and very rare in urinary bladder.[1] The histological character is the presence of xanthoma cells (lipid-laden macrophages), multinucleated giant cells and cholesterol clefts. To authors knowledge, since the first report by Wassiljew in 1932, 37 cases have been reported in the literature. Of the 38 xanthogranulomatous cystitis cases, including the present report, four were children and 34 were adults or adolescence. The median age was 43 years and more female than male (17 cases in males, 20 cases in females, and one unknown) was found.

            Multiple theories regarding the pathogenesis of XC has been proposed, including chronic infection, urachal remnant, retained foreign bodies, immunological disorders, abnormal lipid metabolism, or reaction to a local tumor.[2] 

            In the early year, most of the XC were found located over dome of the bladder and were associated with urachal remnant.[3] With more cases reported, XC are found located in the posterior side or lateral side of bladder, which are not related to urachal remnant. In our review, the location of XC is at dome in 20 of the 38 published cases, and 15 of the cases are associated urachal lesion. Nearly all the reported XC cases are unifocal lesion, but in the present case, three masses are found located at bladder neck, right lateral and left lateral of bladder, respectively. This indicates there is no relationship with an urachus, and has more to do with global condition of the urinary bladder, like infection. The patient has no operation history involving gynecologic or urologic system, and the pathology showed no malignant neoplasm; thus, reaction to retained foreign body or local tumor was less likely.

            The symptoms of XC are unspecific and may be of no help when distinguishing from other bladder diseases. The most common presentation was irritative symptom, including frequency and urgency, and are followed by lower abdominal discomfort and dysuria. Image finding or appearance under cystoscope are often difficult to make difference from urothelial carcinoma, either. Accordingly, the diagnosis of XC is basically based on the pathological result.

            Surgical resection is thought to be the curative treatment.[4] No recurrence has been reported so far. In our review, of the 38 reported cases, 27 cases received partial cystectomy and 7 cases received endoscopic resection of tumor. The use of transurethral resection for XC increased in this decade, this may attribute to the early detection with the image. Noteworthily, conservative treatment was considered to be unwise, and rarely employed previously due to its ineffectiveness. Mahalik reported a 4-year-old boy who presented with acute urinary retention and was diagnosed as XC with pathological evidence.[5]  The boy became asymptomatic after aggressive antibiotics treatment for four weeks. Though lack of long-term follow-up data, this literature discloses the possibility to arrests the disease process in its nascent stages.

            XC is a rare benign chronic inflammatory disease, and should be considered in the differential diagnosis of bladder tumor. The diagnosis is confirmed by histopathological examination. Antibiotics and surgical resection are curative treatment options.

1.         Bates AW, Fegan AW, Baithun SI: Xanthogranulomatous cystitis associated with malignant neoplasms of the bladder. Histopathology 1998, 33(3):212-215.

2.         Yamamoto S, Yoshida K, Tsumura K, Nomiya A, Yoda K, Iida K, Homma Y, Enomoto Y: Xanthogranulomatous Cystitis Treated by Transurethral Resection. Urol Case Rep 2015, 3(5):143-145.

3.         Tan LB, Chiang CP, Huang CH, Chian CH: Xanthogranulomatous cystitis: a case report and review of the literature. Int Urol Nephrol 1994, 26(4):413-417.

4.         Hayashi N, Wada T, Kiyota H, Ueda M, Oishi Y: Xanthogranulomatous cystitis. Int J Urol 2003, 10(9):498-500.

5.         Mahalik SK, Sable MN, Das K: Xanthogranulomatous cystitis masquerading as bladder tumor in a child. Indian J Urol 2020, 36(4):312-314.