Ureteral rupture with retroperitoneal urinoma caused by peritoneal metastases of gastric cancer

Abstract

We describe the rare case of a patient with ureteric rupture during systemic drug treatment for peritoneal metastases of gastric cancer, who underwent double-J stent placement. A 66-year-old man with gastric cancer was referred to the authors’ hospital. Esophagogastroduodenoscopy showed an irregular elevated lesion with thickened gastric folds, and biopsy specimens revealed a poorly differentiated adenocarcinoma. Abdominal contrast-enhanced computed tomography (CT) revealed extensive wall thickening with homogeneous enhancement of the stomach, enlarged lymph nodes in the perigastric area, and nodules in the peritoneal cavity, suggesting peritoneal metastases. The clinical diagnosis was cT4N2M1 with peritoneal metastases, and the patient received chemotherapy (S-1 plus oxaliplatin). After six courses of chemotherapy, the patient presented to the emergency outpatient department with a complaint of acute severe pain in the left lower back. Emergency abdominal contrast-enhanced CT showed extravasation of the contrast medium from the left upper ureter in the periureter area along with the retroperitoneum, and there was no mass lesion or stone in the kidney, ureter, or bladder. A double-J stent was placed under cystoscopic guidance, and no resistance was felt when the stent was inserted. The patient’s postprocedural course was uneventful, and he received ramucirumab in combination with paclitaxel after double-J stent placement. However, 2 months later, systemic drug treatment was discontinued because of loss of appetite and increased general fatigue and changed to the best supportive care. His general condition gradually deteriorated, and he died 3 months after the ureteral rupture. Prompt interventions, including retrograde placement of ureteral stents with concurrent use of antibiotics, will reduce mortality and morbidity in this rare entity.

Introduction

Ureteral perforation, which results in urine extravasation from the ureter, is a rare entity that is usually associated with ureteral obstruction due to nephrolithiasis [1]. Other causes include trauma, pregnancy, posterior urethral valves, vascular extrinsic compression, iatrogenic or idiopathic causes, and invasive urologic procedures; however, spontaneous ureteral rupture is an extremely rare condition [2–4]. Urinoma or abscess formation caused by extravasation of urine may occur, eventually leading to sepsis and death.

Gastric cancer is the fifth most common cancer and third leading cause of cancer-associated deaths worldwide [5]. The peritoneum is considered the most common site for metastasis from gastric cancer, which is mostly caused by the seeding of cancer cells through the peritoneum. Metastasis to the peritoneum or urinary tract may result in renal dysfunction, infection, and painful hydronephrosis, although rupture of the urinary collecting system is rare.

There are only a few published cases of ureteral rupture caused by tumors, which is a rare entity that is reported in the literature in the form of single case reports. Therefore, the optimal management of such conditions remains unclear [6]. Herein, we describe a case of spontaneous ureteral rupture during systemic drug treatment of unresectable gastric cancer treated with double-J stent placement.

Case presentation

All procedures were performed in accordance with the ethical standards laid down in the 1964 Declaration of Helsinki and its later amendments. Informed consent was obtained from the patient for being included in the study.

A 66-year-old man with complaints of loss of appetite and body weight presented to our department for further evaluation after a diagnosis of gastric cancer at his local hospital. Esophagogastroduodenoscopy showed an irregular elevated lesion with thickened gastric folds, edematous changes, erosions in the entire gastric body, and severe luminal narrowing (Fig. 1A). Biopsy specimens of the lesion revealed a poorly differentiated adenocarcinoma, and immunohistochemical analysis of the tumor showed no reactivity to human epidermal growth factor receptor 2. Abdominal contrast-enhanced computed tomography (CT) revealed extensive wall thickening with homogeneous enhancement of the stomach, enlarged lymph nodes in the perigastric area, and nodules in the peritoneal cavity, suggesting peritoneal metastases (Fig. 1B). Upper gastrointestinal fluoroscopy using a contrast agent showed deformity of the gastric contour with poor extension.

Fig. 1.

Esophagogastroduodenoscopy photograph and abdominal contrast-enhanced computed tomography imaging scan. Esophagogastroduodenoscopy shows an obviously elevated lesion with thickened gastric folds, edematous changes, and erosions in the entire stomach (A). Abdominal contrast-enhanced computed tomography shows thickening of the gastric wall with homogeneous enhancement of the stomach and enlarged lymph nodes in the perigastric area (B, arrow)

The clinical diagnosis was cT4N2M1 with peritoneal metastases according to the Japanese classification system of gastric carcinoma [7]. The patient received chemotherapy using S-1 plus oxaliplatin. An 80 mg/m²/day dose of S-1 was administered orally twice daily for the first 2 weeks of the 3-week cycle. On day 1 of the 21-day cycle, the patient received 100 mg/m² of intravenous oxaliplatin, followed by 80 mg/m²/kg of S-1. After six courses of chemotherapy, the patient presented to the emergency outpatient department with a complaint of acute severe pain in the left lower back. Upon examination, the patient was alert, with blood pressure, heart rate, and body temperature measurements of 185/125 mmHg, 80 beats per minute, and 36.9 °C, respectively. Results of his blood analysis were as follows: a decreased red blood cell count (309 × 10⁴/mm³; normal range, 435–555 × 10⁴/mm³), normal white blood cell count (6.8 × 10³/mm³; normal range, 3.3–8.6 × 10³/mm³), normal creatinine level (0.82 mg/dL; normal range, 0.65–1.07 mg/dL), and high C-reactive protein level (0.33 mg/dL; normal range, < 0.14 mg/dL). High levels of the serum tumor marker carbohydrate antigen (CA) 19–9 (169.0 U/mL; normal range, < 37 U/mL), high levels of CA125 (137.0 U/mL; normal range, < 35 U/mL), and CA72-4 (6.2 U/mL; normal range, < 5.3 U/mL) were detected, while the level of the serum tumor marker carcinoembryonic antigen was within the normal range.

Emergency abdominal contrast-enhanced CT was performed. Extravasation of the contrast medium from the left upper ureter in the periureter area along with the retroperitoneum (Fig. 2A, B) revealed no mass lesion or stone in the kidney, ureter, and bladder. A double-J stent was placed under cystoscopic guidance to derivate urine along with the administration of intravenous antibiotics, and no resistance was felt when the stent was inserted (Fig. 3).

Fig. 2.

Abdominal contrast-enhanced computed tomography (CE-CT) scans showing the ureteral rupture. CE-CT in the early phase after injection of the contrast medium showing a dilated left upper ureter with fluid collection in the periureter area and retroperitoneum (A). CE-CT in the delayed excretory phase shows extravasation of the contrast medium around the dilated left upper ureter (B)

Fig. 3.

Double-J ureteral stent insertion under fluoroscopic guidance. Primary ureteroscopy to set the double-J ureteral stent is successfully positioned

The patient’s postprocedural course was uneventful, and his symptoms and overall condition promptly improved. A follow-up abdominal contrast-enhanced CT 9 days later showed a decrease in the size of the retroperitoneal fluid collection (Fig. 4), and the patient was discharged 10 days after ureter stent placement with no further antibiotics. He received ramucirumab in combination with paclitaxel as second-line systemic drug treatment. However, 2 months after double-J stent placement, systemic drug treatment was discontinued because of loss of appetite and increased general fatigue and changed to the best supportive care. His general condition gradually deteriorated, and he died 3 months after ureteral rupture.

Fig. 4.

Abdominal contrast-enhanced computed tomography (CE-CT) scan 9 days after ureteral stent placement. A stent in the left ureter is seen, and fluid collection in the periureter area and retroperitoneum is decreased (arrow)

Discussion

Here, we described the case of a patient who experienced ureteral rupture during chemotherapy for advanced metastatic gastric cancer. Although the optimal management of malignant extrinsic ureteral rupture remains unclear owing to its rarity, ureteral stenting was effective in improving our patient’s condition. We searched the Medline and PubMed databases for English-language literature published between 2000 and 2021 for reports of similar cases using the keywords “tumor” and “ureteral rupture.” We obtained data on age, sex, the primary tumor, treatments, and outcomes for each patient. To our best knowledge, this is the first report of ureteral rupture during chemotherapy for advanced gastric cancer in the English literature. In addition, we searched the Japan Centra Revuo Medicina databases for Japanese-language literature published between 2000 and 2021 for reports of similar cases using the keywords “gastric cancer” and “ureteral rupture.”

The clinicopathological features of the nine previously reported cases of rupture in the urinary collecting system [2, 6, 8–14] and the present case are presented in Table 1. Based on these cases, we found that the median patient age was 66 years (range 28–82 years), and this cohort comprised six men and four women. The primary malignancies leading to rupture of the urinary tract were as follows: two cases of prostate cancer (20.0%), two cases of uterine cervical cancer (20.0%), two cases of bladder cancer (20.0%), two case of gastric cancer (20.0%), one case of ovarian cancer (10.0%), and one case of a desmoid tumor (10.0%). The presenting symptoms of rupture of the urinary tract varied and ranged from abdominal pain, lack of urine passage, back pain, and difficulty urinating. The sites of rupture in the urinary tract were the ureter in four cases, renal fornix in two cases, renal pelvis in two cases, renal calyx in one case, and ureteropelvic junction in one case. Antibiotic treatment was provided in all cases, and treatment consisted of stent placement in six patients, nephrostomy in three patients, and nephrostomy following stent placement in one patient.

Table 1.

Characteristics of the reported cases of rupture in the urinary collecting system caused by a tumor

Author	Year of publication	Patient age (years)	Patient sex	Primary tumor	Symptom	Location of the ureteral rupture	Treatment	Outcome
Cormio G. et al. [2]	2001	66	Female	Ovarian cancer	Right flank pain	Right lower renal calyx	Nephrostomy	Died 1 month after rupture
Singh I. et al. [8]	2009	35	Female	Uterine cervical cancer	Abdominal pain	Right renal fornix	Nephrostomy	ND
Jou Y.C. et al. [9]	2012	77	Male	Bladder cancer	Lack of urine passage	Left renal pelvis	Stent placement	Died 6 months after rupture
Sugiura S. et al. [10]	2013	37	Female	Gastric cancer	Left flank pain	Left renal pelvis	Stent placement	Died 11 months after rupture
Yoon P.D. et al. [11]	2014	28	Male	Desmoid tumor	Abdominal pain	Right lower ureter	Nephrostomy following stent placement	Alive 6 months after rupture
Ikeda S. et al. [12]	2015	66	Female	Uterine cervical cancer	Abdominal pain	Right upper ureter	Stent placement	ND
Deng F. et al. [13]	2015	68	Male	Prostatic cancer	Left flank pain	Left upper ureter	Nephrostomy	Alive 1 month after rupture
Larrache Y. et al. [6]	2020	82	Male	Bladder cancer	Left low back pain	Left renal fornix	Stent placement	ND
Naito M. et al. [14]	2020	67	Male	Prostatic cancer	Difficulty urinating	Left ureteropelvic junction	Stent placement	ND
Present case	2022	66	Male	Gastric cancer	Abdominal pain	Left upper ureter	Stent placement	Died 3 months after rupture

ND not described

Rupture of the ureter is secondary to overpressure exerted on the ureter, which is responsible for the progressive appearance of hydronephrosis and inflammation of the urinary collecting system [6, 15, 16]. Infiltration of the ureteral wall by neoplastic cells may occur in various situations, such as ureteral metastasis through lymphatic and/or blood vessels, peritoneal deposits, lymph node metastases, or direct extension [17–20]. In the case of urinary calculi, impaction of stones on the ureteral wall or a downward moving calculus, which may cause erosion and ulceration of the ureteral wall, may lead to ureteral rupture at the distal ureteral obstruction [21]. Peritoneal dissemination is the most common pattern of metastasis and is usually associated with poor prognosis in patients with gastric cancer. It can cause serious conditions, e.g., intestinal obstruction, massive ascites, and ureteral obstruction. [19, 22]. Hydronephrosis in gastric cancer is mainly caused by metastases to the retroperitoneum, para-aortic lymph nodes, ureters, and bladder [19, 23]. The presence of obstructive pyonephrosis could also be secondary to a ureteral lesion, which may eventually lead to ureteric rupture and extravasation of urine.

In the emergency status including the present case, it seems to be hard to confirm the pathological examination of the tissue around the ruptured ureter. Our patient showed nodules in the peritoneal cavity in the contrast-enhanced CT, suggesting peritoneal metastases. Although it remains unclear whether a ureteral rupture is caused by tumor infiltration or tumor necrosis after chemotherapy in the present case, the latter possibility might be unlikely, because CT images did not disclose any evidence of tumor shrinkage after chemotherapy.

Urinary tract rupture can occur at any level of the urinary collecting system, from the renal calyces to the lower ureter, because the pathogenesis underlying urinary collecting system disruption is believed to be an increase in intraluminal pressure due to obstruction. In our review of ureteral rupture caused by a tumor, the site of rupture was the ureter in four cases and the upper urinary tract in five cases. The most commonly described phenomenon is forniceal rupture, but renal pelvic rupture without forniceal rupture is extremely rare [24]. In most frequent cases of rupture caused by ureteral calculi, one possibility is that smaller distal stones create higher intraureteral pressures than larger proximal stones because of the greater length of the obstructed ureter and smaller diameter of the distal ureter [15].

Although spontaneous ureteral rupture may not always be symptomatic, it can lead to severe complications due to sepsis caused by extravasation of urine, which can lead to morbidity and mortality. Therefore, treatment is based on antibiotic therapy to prevent urinoma. Additionally, ureteral stent placement is the first choice in cases of ureteral rupture, which is also often applied in ureteral obstruction secondary to malignancies. Percutaneous nephrostomy is indicated in cases where ureteral stent insertion is difficult or where the ureteral stent is not effective due to extensive peritoneal metastasis [23]. It is important to assess the patient’s quality of life and survival time because most of these patients are incurable at the time of diagnosis and have a poor prognosis.

According to Japanese gastric cancer treatment guidelines, systemic drug treatment using anti-cancer agents and/or molecular targeted agents is recommended for patients with unresectable advanced and recurrent gastric cancer, an Eastern Cooperative Oncology Group performance status of 0–2, and preserved major organ function [25]. Limited reports suggested a potential survival benefit of chemotherapy in patients with ureteral obstruction secondary to gastric cancer. Migita et al. reported that the median survival was 11.2 months in patients with ureteral obstruction secondary to gastric cancer who received chemotherapy compared to 3.1 months in patients who did not receive chemotherapy (P = 0.0002) [23]. Matsuura et al. identified gastric cancer as the primary disease, a poor performance status before stenting, and best supportive care as treatment after stent insertion as independent prognostic factors in the evaluation of 93 patients with malignant extrinsic ureteral obstruction [26]. Because the prognosis of patients diagnosed with rupture of the urinary collecting system is consistently poor, prompt palliative urinary diversion and subsequent appropriate systemic drug treatment depending on the situation may result in a survival benefit.

Conclusion

Ureteral rupture associated with gastric cancer metastasis, which is an unusual phenomenon that occurs due to ureteral obstruction, was investigated. The prognosis of patients with ureteral rupture due to malignancy is extremely poor, and ureteral stent placement is an effective treatment in the management of this condition. When ureteral rupture occurs, prompt identification and characterization of this condition are necessary to allow equally rapid and appropriate management of this disease. Although prospective studies and/or randomized controlled trials are unrealistic owing to the small number of patients with this disease, further accumulation of data, including case–control studies, is needed to determine the management of such patients.

Author contributions

All listed authors contributed to the original manuscript. TN and MK: designed the study. TN, KY, MM, SU, HM, HK, TT, TK, KI and KH: have been involved in drafting the manuscript and revising it critically for important intellectual content. TN and KH: finalized the manuscript and submitted the paper for publication. All authors have read and approved the manuscript of this case report.

Funding

This case report was not supported by any funding.

Data availability

The data include individual patient data, but the data are available from the corresponding authors upon reasonable request.

Declarations

Conflicts of interest

All authors declare that they have no conflict of interest.

Ethical approval

All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional research committee. For this type of study, formal consent is not required.

Consent for publication

Written informed consent was obtained from the patient for publication of this case report and any accompanying images.