曾世一, 溫聖辰

高雄醫學大學附設中和紀念醫院 泌尿部

Shih-Yi Tseng, Carl Sheng-Chen Wen

Department of Urology, Kaohsiung Medical University Hospital, Kaohsiung, Taiwan

 

Introduction: Xanthogranulomatous cystitis (XC) is a rare, benign chronic inflammatory disorder of unknown etiology that was first described in 1932 [1]. To our knowledge, only a few case reports of XC have been reported in the past literature. XC is characterized histologically by the presence of xanthoma cells (lipid-laden macrophages), multinucleated giant cells and cholesterol clefts. This change has been reported in many organs, such as the colon, ovary, pancreas, salivary gland, appendix, gall bladder, endometrium, brain and kidney [2]. However, it is rarely reported in the urinary bladder. We report a case of XC and share our therapeutic plan.

Case reports: A 36-year-old woman was referred from local medical doctor for management of painless gross hematuria. Tracing back to her medical history, she ever underwent laparoscopic appendectomy 2 years prior admission due to acute appendicitis. Physical examination revealed 3 trocar wound, healing well, were respectively at the umbilicus, left lower quadrant lateral to the rectus muscle, and the suprapubic area. Neither costovertebral angle knocking pain nor low abdominal pain was noted. Urinary cytology and blood biochemical evaluations were unremarkable.

  Abdominal computed tomography (CT) scan on admission revealed one ovid cystic lesion with enhanced thickened wall abutting the right anterior aspect of urinary bladder (fig 1A). Rigid cystoscopy was arranged and showed a huge protruding, polypoidal tumor with necrosis and bleeding surface at anterior wall of urinary bladder (fig 1B). Therefore, we performed laparoscopic partial cystectomy for the patient. Under general anesthesia, the patient was placed on lithotomy position. We implanted ureteral catheter to protect bilateral ureter, and then we used resectoscope to mark the margin of bladder tumor. Then laparoscopic approach (transperitoneal route) was performed, and we found bladder tumor that adhered to the suprapubic abdominal wall (fig 1C). Much to our surprise, the tumor adhesion region was almost below the suprapubic trocar wound ! Due to unobvious tumor margin under laparoscopic field, we simultaneously used light sourse of resectoscope to identify the tumor margin (fig 1D). The pathological report showed the tumor was composed of xanthogranulomatous inflammation. The postoperative recovery was uneventful and she was discharged home on postoperative day 6th. At 3 months post-resection, there was no evidence of recurrence adjacent to the previous resection scar.

Discussion: Xanthogranulomatous cystitis is a rare inflammatory disorder. Multiple theories about its etiology have been reported, including anaerobic urinary tract infection, presence of suture material, urachal adenoma, and malignancy [3]. However, due to the bladder tumor was located below the previous trocar wound, iatrogenic xanthogranulomatous cystitis was highly suspected in this case. About xanthogranulomatous cystitis, the curative treatment of choice is surgical resection [4]. Generally, simple tumor excision was suggested to treat localized disease. However, partial cystectomy will be preferred when the tumor is combined with urachal remnant or adenoma [4]. The treatment of this case is remedied by simple excision of the lesion and a close follow-up. Timely diagnosis and early intervention are the key to treat this rare disease.