Korean J Urol Oncol 2011;9(1):35-38 Recurred Localized Amyloidosis of Bladder Causing Hematuria Dong Gyu Jang1, Sun Ju Lee1, Jongkeun Kim1, Ji-Won Hwang1, Changhee Yoo1, 1 2 1 Cheol Young Oh , Sun Young Jun , Jin Seon Cho Departments of 1Urology, 2Pathology, College of Medicine, Hallym University, Chuncheon, Korea Localized amyloidosis of the bladder is an uncommon disease. As it can be mistaken as a malignant disease, urinary bladder neoplasms should be included as differential diagnosis. And systemic amyloidosis also should be considered because the prognosis of localized amyloidosis is very different from that of systemic amyoidosis. The primary treatment for localized amyloidosis of the bladder is transurethral resection of the mass. And annual cystoscopic evaluation is recommended to detect recurrence. In the current case, the patient underwent transurethral resection of bladder mass twice for 2 years, and supposed to be recurred amyloidosis due to proliferation of residual amyloid. Because localized amylodosis of bladder can be recurred, long-term follow-up is needed after complete resection of the mass. (Korean J Urol Oncol 2011;9:35-38) Key Words: Amyloidosis, Hematuria, Urinary bladder neoplasms Amyloidosis is a disease characterized by extracellular deposi- Urinalysis showed many RBC/HPF, and urinary pH was 5.5. tion of the fibrillar protein, called ‘amyloid'. Deposition of No urinary glucose or white blood cells were detected. No ma- amyloid can be localized or systemic. Primary localized bladder lignant cells were detected in voided urine cytology and her amyloidosis is a rare condition. Clinically, it is difficult to dis- excretory urography showed no abnormal finding. A computed tinguish the localized bladder amyloidosis from urinary bladder tomograqphy (CT) scan revealed focal asymmetric wall thick- neoplasms due to the similarity of symptoms, and cystoscopic ening of left side of bladder base (Fig. 1). Cystoscopic findings and radiologic findings. showed diffuse erythematous and bullous mucosal lesions on trigone and left lateral wall surrounded by normal mucosa We report a case of recurrent primary amyloidosis of urinary (Fig. 2). Transurethral resection of the bladder lesions (TURB) bladder. performed. On the microscopic examination, nodular deposits of amorphous eosinophilic material were seen in the lamina CASE REPORT propria and the vascular wall (Fig. 3). Microscopic calcificaA 71-year-old woman came to the urology department with tions and foreign body giant cell reactions were also seen ad- complaints of intermittent, painless gross hematuria for 3 jacent to the deposits. On Congo red amyloid staining, the de- months dysuria and residual urine sense for recent 5 days. Her posits exhibited mahogany red color (Fig. 4). Those deposits medical history was unremarkable. And she had not smoked showed apple green birefringence under a polarized microcope, or drunken alcohol at all. consistent with amyoloidosis (Fig. 5). After 2 months of operation, the patient revisited urology de- Received March 16, 2011, Revised April 11, 2011, Accepted April 11, 2011 Corresponding Author: Jin Seon Cho, Department of Urology, Hallym University Sacred Heart Hospital, 896, Pyeongchon-dong, Dongan-gu, Anyang 431-070, Korea. Tel: 82-31-380-3851, Fax: 82-31-380-3852, E-mail: [email protected] partment with 3-days total painless gross hematuria. On cystoscopy, we found unhealed scar tissue and remnant erythematous mucosal lesion around previous operation site. Again, the result of voided urine cytology was negative for malignancy. Since hematuria and voiding symptoms disappeared spontaneously, 35 36 대한비뇨기종양학회지:제 9 권 제 1 호 2011 the patient was recommended follow-up OPD without treat- finding was detected in cystoscopy and serial urine cytology. ment. Then, she came to the urology OPD again for gross hem- Because the symptoms improved spontaneously at this time, the aturia after 1 year. Cystoscopic examination demonstrated a sol- patient has been on routine follow-up schedule with con- id broad-based mass like lesion with an edematous and smooth servative treatment. surface on the left lateral wall and on the trigone near the bladder neck. The results of urine cytology and CT scan were DISCUSSION unremarkable. After TURB, the histologic examination revealed recurred amyloidosis. Systemic evaluations including electro- Amyloidosis can be classified into systemic (multiple myelo- cardiogram, colonoscopy with rectal biopsy, abdominal fat bi- ma, chronic inflammatory conditions and chronic hemodial- opsy, serum protein electrophoresis, 24hr urine electrophoresis, ysis), hereditary (familial mediterranean fever and familial amy- free light chain assay, echocardiography, chest CT, and nerve loidotic neuropathies), localized and idiopathic forms.1 conduction study, were performed to exclude systemic amyloi- The etiology of this disorder is unknown, but some hypoth- dosis, and no abnormal finding was detected. Finally, the bladder mass was diagnosed as a localized bladder amyloidosis. She visited the clinic again with complaints of recurrent gross hematuria in 3 monthes after 2nd TURB. No abnormal Fig. 1. Focal asymmetric wall thickening on the left posterior bladder wall without definite enhancement in the thickened wall. Fig. 2. Diffuse lesions on trigone and left lateral wall with normal surrounding mucosa. Fig. 3. Amorphous eosinophilic materials are deposited in the lamina propria and the vascular wall (H&E, A: x40, B: x100). Dong Gyu Jang, et al:Recurred Localized Amyloidosis of Bladder Causing Hematuria 37 Fig. 4. The deposits exhibit mahogany red color with Congo red amyloid staining (x100). Fig. 5. Apple green birefringence under a polarized microscope, consistent with amyoloidosis. esis suggest that amyloidosis can be related to recurrent, chron- recurrence. ic inflammation and migration of lymphoplasmacytic cells to 2 Tirzman et al. followed up 24 patients with localized bladder amyloidosis from 2 to 36 years; 25% remained disease free, the site. Relevant to the genitourinary amyloidosis, involvement of 21% had stable lesions and 54% recurred.8 In our case, the pa- the kidney, ureters, seminal vesicles, prostate, penis, and testis tient has been followed up for 2 years. After 1 year of the first has been reported. More than half of the localized genitourinary TURB, amyloidosis recurred in the bladder and the second 3 amyloidosis is located in the urinary bladder. Primary amyloidosis of the bladder is rare, and an initial examination may be easily confused with urothelial carcinoma TURB was done. It seems that recurrent hematuria has been caused by proliferation of residual amyoid and, if possible, complete resection and long term follow up is needed. due to a similar clinical presentation of painless gross hematuria. 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