輸尿管膀胱重建術後之反覆氣腫性腎盂炎罕見案例

洪紹綸¹、吳俊賢^1,2、林嘉祥^1,2

¹義大醫療財團法人義大醫院 泌尿科；

²義守大學 醫學系

A Rare Case of Recurrent Emphysematous Pyelitis after Ureteroneocystostomy

Leo Shao-Lun Hung¹, Chun-Hsien Wu^1,2, Victor C. Lin^1,2

¹Department of Urology, E-Da Hospital, Kaohsiung, Taiwan;

²School of Medicine, College of Medicine, I-Shou University, Kaohsiung, Taiwan

Abstract

Emphysematous pyelitis (EP) is a rare infection disorder characterized by gas formation confined to the collecting system. In contrast, emphysematous pyelonephritis (EPN), presenting with more severe clinical symptoms, involves gas retention in the renal parenchyma and/or perinephric areas and is more familiar to urologists. If not treated early, EP may progress to EPN, requiring more intensive treatment. We present a case of recurrent EP with a complex medical history, unique findings on abdominal plain film, and the treatment course.

A 65-year-old female with a history of endometrial stromal sarcoma post radical hysterectomy, partial cystectomy, left ureteroneocystostomy, and adjuvant radiotherapy 20 years ago, presented with recurrent urinary tract infection and bilateral hydronephrosis during the computed tomography (CT) follow-up. She denied obvious flank pain and was initially managed with oral antibiotic therapy A videourodynamic study (VUDS) revealed small bladder capacity and poor bladder compliance without visible vesicoureteral reflux (VUR) bilaterally. Clean intermittent self-catheterization (CISC) was instructed with residual urine volume around 150-200 ml.

However, pneumaturia was noted subsequently and a follow-up abdominal CT identified gas in the left upper urinary tract, suggestive of emphysematous pyelitis. Left antegrade double-J stent insertion was performed with temporarily resolution of left EP. However, gas retention over left collecting system recurred three months later after removal of the left ureteral stent. She was admitted for left ureterorenoscopy (URS) examination, and mild ureteroneocystostomy stenosis was noticed post laser ureterotomy.

Diagnostic considerations included left VUR secondary to poor bladder compliance and ureteroneocystostomy, air introduction during CISC and a possible ureteral stricture with infection-related gas production. Operative findings confirmed a neo-ureteral orifice stenosis, contributing to recurrent infections and gas formation. Temporary resolution of EP and infection was achieved with ureteral catheterization. However, the cause of EP remains uncertain and needs further studying.

This case highlights the challenges of managing emphysematous pyelitis in the context of complex urological history. The interplay of poor bladder compliance, ureteral stricture, and infection created a multifactorial scenario for recurrent gas formation. Early identification and resolution of obstructions, coupled with infection control, are critical to managing this rare condition and preventing recurrence.