腫瘤復發造成主動脈腸道瘻管 - 罕見之上泌尿道尿路上皮癌表現

陳人傑¹、黃子豪^1,2、黃志賢^1,2

¹臺北榮民總醫院 泌尿部; ²國立陽明交通大學醫學院 泌尿學科 書田泌尿科學研究中心

Recurrent tumor causing aorto-enteric fistula – a rare presentation of upper tract urothelial carcinoma

Jen-Chieh Chen¹, Tzu-Hao Huang^1,2, William J. Huang^1,2

¹Department of Urology, Taipei Veterans General Hospital;

²Department of Urology, College of Medicine and Shu-Tien Urological Research Center, National Yang Ming Chiao Tung University, Taipei, Taiwan²

Introduction:

Fistulous communication between the aorta and colon following nephrectomy is extremely rare. Here we report a case of aorto-enteric fistula owing to tumor recurrence after nephroureterectomy, which presented as massive bloody stool and hypovolemic shock and was controlled by angiographic embolization.

Case report:

This is a 76-year-old man with history of hypertension and type 2 diabetes mellitus. He had intermittent gross hematuria for 6 months, accompanied with mild pain over left lower quadrant of abdomen and body weight loss (7 kilograms (10%) in 6 months). He came to our hospital in April 2020. Abdominal ultrasound and computed tomography showed a heterogenous mass at left renal pelvis with hydronephrosis. No metastasis was identified on chest x-ray and whole body bone scan. He then underwent nephroureterectomy with bladder cuff excision in June 2020. Locally advanced tumor involving renal hilum, and perirenal fat was noted during surgery (R2 resection). Pathology reported invasive high grade urothelial carcinoma (pT4) and sarcomatoid differentiation. Expression of programmed death-ligand 1 (PD-L1) was positive for SP-142(IC 30%). Of note, the patient initially had marked anemia (hemoglobin 5.7) and leukocytosis (WBC 45000), which were ultimately determined as tumor associated leukemoid reaction, significantly improved within days postoperatively.

One month after surgery, chest CT surprisingly disclosed a 7.6 cm heterogenous mass at left retroperitoneum, suggesting the presence of local recurrence. In the meantime, the patient had a rapid decline in general performance condition, making him ineligible for chemotherapy. Thus atezolizumab and palliative radiotherapy were administered (about 6 weeks after surgery). Unexpectedly, the patient stabilized after 1^st course of atezolizumab and showed a dramatic improvement in his general condition. He then underwent regular atezolizumab administration every 3 weeks for 6 courses uneventfully until November 2020.

During hospitalization for the seventh cycle of atezolizumab, hypotension with increased heart rate was found several hours after drug administration, then massive dark hematochezia was noted. Hypovolemic shock soon developed, and we started aggressive resuscitation as well as intubation for unstable vital signs. Colorectal consultation suggested image evaluation with embolization. CT scan surprisingly revealed an active bleeder at left renal artery stump, showing contrast leakage into the recurrent tumor and descending colon, which literally became an aorto-enteric fistula and misled us into believing that the whole scenario was a profound gastrointestinal bleeding. He was immediately brought to angiography room and the radiologist embolized the artery stump successfully. He totally received more than 50 units of blood products within 6 hours, as the vital signs were extremely unstable throughout the procedures. Afterward, he was transferred to intensive care unit. Condition stabilized under supportive care, and he was transferred to ordinary ward after one week. All vital life supportive facilities were discontinued, as he had relatively good appetite and smooth oral intake. By that time, we had been speaking to other specialists and trying to arrange further treatment to prevent possible deterioration.

Unfortunately, loss of consciousness with cardiac arrest occurred after 2 days back to the ordinary ward. Massive bleeding and clots from anus were found, which was exactly the same scenario as the last episode. We informed his family of his critical condition and poor prognosis, and the patient finally expired due to hypovolemic shock.

Conclusions:

There is no prior case report describing a fistulous communication between the renal artery stump, which actually was the aorta, and colon owing to tumor recurrence after nephroureterectomy. If the whole event had not occurred during hospitalization, the patient might have died of massive bleeding directly, without having the chance to be rescued and being correctly diagnosed. In this case, the patient was treated successfully with angiographic embolization initially. However, rapid recurrent bleeding as a result of tumor aggressive invasion occurred out of our expectation. Placement of covered stents to the lesion might be the solution in this case and could have been performed right after the first bleeding episode to prevent future bleeding.