病例報告: 骨盆腔手術及放射治療後產生的雙側輸尿管動脈廔管

黃昱凱¹, 黃子豪^1,2,3, 黃志賢^1,2,3

¹泌尿部 台北榮民總醫院

²書田泌尿科學研究中心

 ³國立陽明交通大學醫學院泌尿學科

Case report: Bilateral uretero-arterial fistula after pelvic surgery and radiation therapy

Yu-Kai Huang¹, Tzu-Hao Huang^1,2,3 William JS Huang^1,2,3

¹Department of Urology, Taipei Veterans General Hospital

² Shu-Tien Urological Science Research Centerand Department of Urology

³ School of Medicine, National Yang Ming Chiao Tung University, Taipei, Taiwan

Purpose:

Uretero-arterial fistula (UAF) is a rare but life-threatening condition typically manifesting with gross hematuria and flank pain. It involves an abnormal connection between arterial vessels and the ureter, commonly occurring at the crossing of the ureter and the common iliac artery. Risk factors for secondary fistula formation include chronic indwelling ureteral stents, pelvic surgery, and radiation therapy, which can lead to inflammatory and devascularization damage to the ureter. Given the rarity and complexity of this condition, understanding its diagnosis and management is crucial.

Case presentation:

A 62-year-old female with history of cervical squamous cell carcinoma, pT1b1N0M0, who underwent radical abdominal hysterectomy (RAH), bilateral salpingo-oophorectomy (BSO), pelvic lymph node dissection (PLND), and para-aortic lymph node sampling (PALNS) , followed by concurrent chemo-radiotherapy (CCRT). The chemotherapy regimen was composed of 6 cycles of Cisplatin and Topotecan, and radiotherapy was 6760cGy/30Fx for gross recurrent tumor over vaginal cuff and 5040cGy/28Fx for pelvis. Then, she presented with bilateral hydronephrosis and hydroureter necessitating bilateral double-J ureteral stent insertion one year after CCRT.

Three years after CCRT, she experienced severe septic shock with gross hematuria, revealing a hematoma in the right renal pelvis on CT angiography and a 0.6cm saccular aneurysm at the right external iliac artery on intra-arterial digital subtraction angiography (IA DSA). 8mm x 5cm stent graft was placed at proximal external iliac artery due to suspected right uretero-arterial fistula. However, a month later, gross hematuria recurred, leading to the diagnosis of a newly occurred left uretero-arterial fistula. A 8mm x 13.5mm vascular plug was deployed at left internal iliac artery distal to the fistula and the proximal left internal iliac artery was embolized by coils. She was once admitted to ICU and temporarily on hemodialysis as a result of repeated major bleeding and contrast medium administration. Ultimately, she was discharged under relative stable condition.

Unfortunately, another episode of gross hematuria and right flank pain occurred after one year in 2023. Again, IA DSA revealed suspicious extravasation at right side proximal external iliac artery. We embolized the right internal iliac artery with a 12mm type 2 vascular plug to prevent backdoor flow and a 13mm x 5cm stent graft was implanted from right external iliac artery to common iliac artery. The symptoms resolved and the patient was discharged in a stable condition after observation.

Discussion:

Reviewing literature on UAF reveals its association with pelvic surgery, radiation therapy, and chronic ureteral stenting, with gross hematuria being the predominant clinical presentation. Diagnostic modalities include abdominal and pelvic angiography, although some cases may fail to indicate the existence of a fistula. The diagnosis was challenging given its rarity as a cause of gross hematuria, with bilateral involvement being exceptionally unusual. Commonly involved arteries include the common iliac and external iliac arteries, with fewer occurrences in the internal iliac artery. Endovascular interventions, such as graft stent placement, are frequently employed. Our case underscores the importance of considering UAF in patients with relevant medical histories and highlights the need for standardized management protocols to optimize patient outcomes.