Journal of Iranian Medical Council

Journal of Iranian Medical Council

Spontaneous Extraperitoneal Bladder Wall Rupture Due to Emphysematous Cystitis: A Case Report and Literature Review

Document Type : Case report

Authors
Department of Urology, Shariati Hospital, Faculty of Medicine, Tehran University of Medical Sciences, Tehran, Iran
Abstract
Abstract 
Emphysematous cystitis is a rare condition that can have fatal complications if left untreated. A timely diagnosis and proper treatment are crucial to achieving appropriate outcomes. Patients with this condition should be evaluated, and if necessary, conservative treatment or surgery should be provided. This case report describes a 73-year-old male patient with emphysematous cystitis who underwent conservative treatment for bladder rupture and reviews the existing literature on this topic. The gold standard for diagnosis of EC is a computerized tomographic scan. This condition should be evaluated, and if necessary, conservative treatment or surgery should be provided.

Keywords

Subjects


Abstract 
Emphysematous cystitis is a rare condition that can have fatal complications if left untreated. A timely diagnosis and proper treatment are crucial to achieving appropriate outcomes. Patients with this condition should be evaluated, and if necessary, conservative treatment or surgery should be provided. This case report describes a 73-year-old male patient with emphysematous cystitis who underwent conservative treatment for bladder rupture and reviews the existing literature on this topic. The gold standard for diagnosis of EC is a computerized tomographic scan. This condition should be evaluated, and if necessary, conservative treatment or surgery should be provided.
Keywords: Abdominal injuries, Aged, Conservative treatment, Cystitis, Humans, Male, Tomography, Urinary bladder, X-Ray computed

Introduction
Emphysematous Cystitis (EC) is an uncommon condition that mostly occurs in elderly female patients with diabetic cystopathy, immunodeficiency, bladder outlet obstruction, neurogenic bladder, recurrent urinary tract infections, or transplanted organs (1).  The gold standard for diagnosing this condition is the Computerized Tomographic (CT) scan (2). It is diagnosed based on the presence of gas in the bladder wall and lumen. Spontaneous rupture of the bladder wall can occur rarely as a complication of EC. If it is not diagnosed and treated promptly, it can lead to peritonitis, sepsis, and eventually death (1). This condition is usually managed with antibiotic therapy or surgery (3). However, recent reports and international guidelines suggest that conservative treatment could be an option in extraperitoneal ruptures (4,5). Since there have been only scant case reports regarding bladder rupture in EC patients, management and treatment of this condition is remains underexplored with limited guidance. In this case report, a 73-year-old male patient with bladder rupture secondary to EC is presented and different treatment options based on a literature review and experience are discussed. Written informed consent was obtained from the patient for publication of this report.

Case Presentation
A 73-year-old male patient presented to the emergency room complaining of hematuria for one day. Other significant symptoms reported by the patient included vague pain in the lower abdomen and severe dysuria, urgency, and frequency. Additionally, he experienced obstructive lower urinary tract symptoms such as intermittency and post-voiding dribbling. The patient had a medical history of Diabetes Mellitus (DM), Benign Prostate Enlargement (BPE), and hypertension. Furthermore, he had been hospitalized for 36 days due to COVID-19 and had been discharged four days earlier. To manage his COVID-19, the patient had been prescribed 25 mg of prednisolone daily, which was gradually tapered. Additionally, he took aspirin 80 mg daily to manage his underlying medical condition. 
In the examinations, the patient was afebrile and had no symptoms of shock. At the time of admission to the hospital, he had a blood pressure of 110/90 mmHg, a pulse rate of 92/min, a respiratory rate of 16/min, and a temperature of 37.4°C. Aside from that, the patient had no costovertebral tenderness, lower abdomen tenderness, rebound tenderness, or guarding. Upon arrival, the laboratory data revealed leukocytosis (white blood cells: 13,700/mm³) and elevated C-reactive protein (CRP: 140 mg/L). Other laboratory results including hemoglobin, platelets, blood urea nitrogen, creatinine, and lactate were unremarkable. Bacteriuria, pyuria, and Hematuria were detected on urinalysis. The abdominopelvic CT scan revealed diffusely increased wall thickness and disruption of the bladder with air bubbles within the bladder wall and the para-vesicle spaces, indicating extraperitoneal dehiscence of the bladder wall (Figure 1).
In light of the patient’s condition and the CT scan results, after taking the urine cultures, ceftriaxone 1 g IV q 12 hr was started, and a catheter was placed for urine drainage. The urine culture revealed Escherichia coli as the cause of the infection and it was sensitive to the third-generation cephalosporin. Seventy-two hours after initiating the antibiotic therapy, the symptoms were improved. A repeat CT scan on day 10 of the treatment (Figure 2) showed that the bladder wall was cured, and the patient was discharged. The patient’s blood sugar was controlled during the follow-up, and an alpha-blocker was started to treat his BPE. The catheter was removed one week after discharge. The patient was followed for six months later, and he had no lower urinary tract symptoms or hematuria during this period.

Table 1. Extra-peritoneal bladder rupture due to emphysematous cystitis cases reported in English literatures have been published on this disease until June 14, 2022

Year/Author

Age

Gender

Predisposing risk factor

Clinical presentation

Urine culture results

Radiologic finding

Management/final outcome

2009/Hu SY 7

82

Female

Pervious history of lung cancer with surgery and radiotherapy

Abdominal pain with tenderness

E. coli

Extra peritoneal perforation

Antibiotic therapy- did not consent to the surgery-

the patient died

2021/Okello C 1

84

Male

Peritoneal dialysis

Hematuria, Voiding symptoms, Fever, Hypotension and Mild suprapubic tenderness

E. coli

Small perforation at the bladder dome

Antibiotic therapy and conservative management-the patient survived

2016/Roels P 5

74

Female

-

Suprapubic pain and progressive deterioration of general condition

E. coli

Extraperitoneal bladder perforation

Antibiotic therapy and conservative management-the patient survived

2012/Qin Y 4

86

Female

Post-operative

Vomiting, Constipation and Lethargy

Klebsiella pneumoniae

Extraperitoneal bladder perforation

Antibiotic therapy and conservative management-the patient survived

2012/Tsubouchi K 8

77

Female

DM-Cerebral infarction

Fever

E. coli, Enterobacter cloacae, Enterococcus faecalis, Enterococcus gallinarum

Extraperitoneal bladder perforation

Antibiotic therapy and surgery-the patient survived

Discussion
Emphysematous cystitis is a rare condition that can be fatal if not diagnosed and treated appropriately in a timely manner (6). Bladder rupture due to EC is an uncommon complication of this disease. In this report, we describe a case of EC with bladder rupture that was managed by urinary drainage and proper antibiotic therapy. A review of the English literature was also conducted using the search terms “emphysematous cystitis” and “bladder rupture” in the title and abstract of articles in PubMed and Google scholar until June 14, 2022. Intra-peritoneal bladder ruptures were excluded. A total of 5 case reports were found and they are summarized in table 1 (1,4,5,7,8).
EC has several known risk factors, including poorly controlled diabetes, bladder outlet obstruction, recurrent urinary tract infections, immunosuppression, female gender, transplant recipients, and conditions causing urinary stasis such as the neurogenic bladder (9). Among the reported cases of bladder rupture due to EC, DM was the most prevalent predisposing factor (43.75%). The aforementioned case had uncontrolled diabetes, BPE-related retention, and prednisolone-related immunosuppression, which predisposed him to the EC. A distinguishing characteristic of this patient was his male gender. Meanwhile, most of the previously reported cases were female (75%). 
The presentation and prognosis of emphysematous cystitis were very diverse (10). Based on the available literature, the most common symptoms were abdominal pain with the highest prevalence, followed by pneumaturia, dysuria, frequency, and urgency (3). Although pneumaturia is one of the first specific symptoms of this disease, it has not been reported in any of the patients who had a ruptured bladder (Table 1), including ours. The aforementioned patient had no pneumaturia and was instead suffering from macroscopic hematuria, which was also present in three of the reported cases (1,11,12). This review showed that abdominal pain (56.25%) and fever (50%) are the most prevalent symptom among these patients. 
Escherichia coli (E. coli) (61.58%) and Klebsiella pneumoniae (23.07%) are the main culprits reported in the previous case reports. There are also reports of patients with positive culture of Enterococcus Faecium, Acinetobacter baumannii, Enterobacter cloacae, Enterococcus faecalis, Enterococcus gallinarum and Candida Albicans (8,11,13). The urine culture of the aforementioned patient was also positive for E-Coli. There is no general agreement available for the treatment of bladder rupture due to emphysematous cystitis. Intraperitoneal ruptures are usually accompanied by acute abdomen or sepsis, requiring surgical repair (5). Some previous studies have recommended managing uncomplicated extraperitoneal ruptures conservatively, similar to other cases of EC, with broad spectrum antibiotics, glycemic control, and correction of underlying comorbid disorders (7,14). Our patient was also treated conservatively without any surgical intervention due to the absence of critical complications from bladder rupture. Although the prognosis is exceptionally diverse, experts believe that the key factors in determining the prognosis in these patients are timely initiation of conventional therapy and the patient’s condition at presentation. Despite the logical conclusion that the prognosis for patients with kidney and ureter involvement requiring surgery would be worse, several guidelines recommend conservative treatment in patients with a small, uncomplicated intraperitoneal rupture of the bladder wall (1,15).

Conclusion 
One of the important manifestations of Emphy-sematous cystitis is bladder rupture, which is a life-threatening condition. It is imperative that these patients be evaluated early and appropriately, and that they receive either conservative treatment or surgery. Patients who have stable conditions and respond adequately to medical treatment may not require surgery.

Consent for publication 
Written informed consent was obtained from the patient for publishing this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal. 

Funding/Support 
This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.

Conflict of Interest 
The authors declare no conflict of interest.

 

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