以膀胱過動症表現之生殖泌尿道外腎源性腺瘤 – 一病例報告與文獻回顧
張孟霖12、傅士彰3、張廷瑞4、林孝友1
1臺北醫學大學附設醫院 泌尿科；2衛生福利部部立基隆醫院 泌尿科；3衛生福利部部立桃園醫院 泌尿科；4台南市立醫院 泌尿科
Extragenitourinary nephrogenic metaplasia / adenoma presenting as overactive bladder syndrome – a case report and literature review
Meng-Lin Chang12, Shih-Chang Fuh3, Ting-Jui Chang4, Hsiao-Yu Lin1
1Department of Urology, Taipei Medical University Hospital, Taipei 110, Taiwan; 2Department of Urology, Keelung Hospital, Ministry of Health and Welfare; 3Department of Urology, Taoyuan Hospital, Ministry of Health and Welfare; 4Department of Urology, Tainan Municipal Hospital
Background:
Overactive bladder (OAB) is a syndrome that causes sudden and unstoppable feeling to urinate. According to International Continence Society (ICS) 2010, OAB is defined as urinary urgency, usually with urinary frequency and nocturia, with or without urgency urinary incontinence. Mainstay treatment of OAB is anticholinergics and beta-3 adrenergic agonist is a new direction of OAB management.
Case Report:
We present a 49-year-old female patient with urgency, frequency, and nocturia. Urinalysis showed no pyuria but microscopic hematuria (WBC 0-2, RBC 2-5). Anticholinergic was prescribed and there was little improvement after two weeks treatment. Suprapubic sonography is performed and showed residual urine 160ml and a 4.3 x 4.1 cm sized cystic mass, which protruded into bladder trigone. She then was transferred to GYN for study the nature of the mass. Per-vaginal examination is normal and trans-vaginal sonography showed no abnormality in vagina, cervix, uterus, except a cystic mass between vagina and bladder. Cystoscopy was performed and showed intact mucosa in urethra, bladder neck, and bladder. Computed tomography showed 4.1 x 4.3 x 3.3 cm sized multi-sepatated cystic space-occupying lesion causing posterior compression of vagina, anterior compression of base of urinary bladder, suspect abscess. Beta iodine was injected into the mass and there was no connection to urinary tract or to vagina. Operative management was carried out in two stages. In stage one, marsupialization was performed. The fluid inside the cystic mass was clear yellowish fluid. Analysis of the fluid showed elevated creatinine, and fluid cytology showed atypical cell. After three weeks, no significant shrinkage happened on sono and we performed total excision of cystic mass through vaginal approach. Carefully dissection was carried out to prevent injury to urethra, bladder neck and bladder base. Foley catheter was placed for two weeks. After removing Foley catheter, the symptoms of urgency, frequency and nocturia were gone and residual urine after voiding decreased to less than 20ml. Pathological finding of excised mass revealed nephrogenic metaplasia /adenoma.
Discussion:
Nephrogenic metaplasia / adenoma is a rare benign lesion and is usually found within genitourinary tract (ureter and bladder). There are also rare case reports that nephrogenic metaplasia / adenoma is found in renal cortical cyst, urethral diverticulum (usually in female). Nephrogenic metaplasia / adenoma is considered to be related with chronic inflammation secondary to genitourinary trauma, prior surgery, renal calculi, or repeated instrumentation. In our case, the tumor is outside urinary tract without connection to bladder or vagina. The mass effects between bladder and vagina may contribute to overactive bladder syndrome. Hence, we recommend initial evaluation of female overactive bladder syndrome should include detailed physical pelvic examination and ultrasound investigation.