Recurrent adult urinary tract infection secondary to unrecognized ureterocele urolithiasis

Abstract

Background: A ureterocele is a cystic dilation of the distal ureter, most commonly presenting as a congenital abnormality that may be associated with other renal anomalies. Ureteroceles present in a variety of manifestations in both children and adults, from asymptomatic to significant flank pain with urolithiasis or recurrent urinary tract infections (UTIs) that often mimic other more common renal pathologies, owing to its challenging clinical diagnosis.

Case Presentation: We report the case of a 78‐year‐old female patient who presented with recurrent UTIs and failure to thrive, complicated by a large stone that first presented in the bladder on computed tomography scan and was later found in the distal right ureter.

Conclusion: This case underscores the varied and often deceptive clinical presentation of ureterocele, stressing the importance of timely diagnosis to prevent secondary complications.

Graphical abstract

Keywords

adult / bladder / ureteral calculus / ureterocele

1 INTRODUCTION

Ureterocele is a congenital cystic dilation of the distal ureter and a rare cause of urinary tract obstruction[1–3]. Ureterocele is a well‐known occurrence in the pediatric urology population but remains a diagnostic and treatment challenge in the adult population. Its prevalence is estimated to be between 1 in 500 and 1 in 4000 patients, and it more commonly affects females, partly due to duplex renal systems presenting more commonly in women[4,5]. The left side tends to be more affected and 10% of cases present bilaterally.

Ureteroceles are classified by location according to the American Academy of Pediatrics as intravesical or extravesical[6,7]. Extravesical ureteroceles are the more common of the two, generally found as ectopic insertions near the bladder neck or urethra and almost always associated with a duplex collecting system[6,8]. Extravesical cases carry the greatest risk of renal tract obstruction due to the potential for prolapse into the bladder neck and for bladder outlet obstruction. Intravesical ureteroceles typically occur at the ureterovesical junction (UVJ) and are more common bilaterally and in adults.

Ureteroceles have a wide variety of clinical presentations, but the most common symptoms range from incontinence, recurrent urinary tract infections (UTIs), urolithiasis, to irregular flank pain. Failure to diagnose and treat ureterocele promptly during childhood may lead to secondary complications in adulthood such as urinary retention, stone formation, chronic renal failure, and urosepsis. In adults, ureteroceles are often clinically silent until the presence of a stone inside the ureterocele, usually solitary, cause flank pain or recurrent UTIs[5]. A large ureterocele that becomes prolapsed may even present as a vaginal mass in females[9]. Ureteroceles are frequently incidental findings on imaging in adults. Understanding the different classifications and variable presentation of ureterocele is crucial for accurate and timely diagnosis thereby preventing complications. This case report highlights the rare incidence of ureteroceles in adult patients and presents a perspective of ureterocele with recurrent UTIs and bladder calculus.

2 CASE PRESENTATION

A 78‐year‐old female with a past medical history of dementia, nonverbal at baseline, noninsulin‐dependent diabetes mellitus, hypertension, hyperlipidemia, recurrent UTIs, and chronic right hydronephrosis presented to the emergency department (ED) with her family due to one month of progressive functional decline, weight loss, poor appetite, and lethargy. The family reported no gross hematuria in her diapers, any discomfort during urination, or any change in her chronic diarrhea. Her most recent UTI was two months prior to presentation at her nursing home with hematuria and urine culture positive for Escherichia coli. The patient was also witnessed to frequently scratch her mons pubis with recent purulent discharge from the skin.

Urinalysis (UA) revealed positive leukocytes and proteinuria. Urine culture showed extended‐spectrum beta‐lactamase (ESBL)‐positive E. coli and Klebsiella pneumoniae. Lactic acid was 4.62 mmol/L on presentation, which quickly corrected to 1.82 mmol/L with intravenous fluids. Blood culture revealed methicillin‐susceptible Staphylococcus species.

Current computed tomography (CT) with intravenous (IV) contrast revealed a large stone, measuring 2.4 cm × 1.9 cm × 1.9 cm, within the right ureteropelvic junction (UPJ) (white arrow in Figure 1A). Additional stones were present in the distal right ureter, the largest being 1.9 cm × 1.3 cm × 1.0 cm in size (white arrow in Figure 1B). No left‐sided renal or ureteral calculus was present. Left‐sided ureterocele was noted (black arrow in Figure 1A). Circumferential bladder wall thickening suggestive of cystitis was also noted.

She had a CT scan of the abdomen and pelvis performed four years prior to current visit, which demonstrated the presence of bladder stones (Figure 2). At the time, she presented to the ED with abdominal pain, vomiting, lethargy, and confusion. An abdominal ultrasound (US) suggested acute cholelithiasis and cholecystitis with right hydronephrosis. The CT scan revealed moderate right hydroureteronephrosis due to a 2.0 cm × 0.9 cm urinary bladder stone projecting into the UVJ and mild left hydroureteronephrosis without obstruction. She underwent endoscopic retrograde cholangiopancreatography to rule out choledocholithiasis and a subsequent laparoscopic cholecystectomy. Her hydronephrosis was managed with conservative observation.

At her current presentation, she began immediate treatment with meropenem given her history of ESBL E. coli and vancomycin for soft tissue infection on her mons pubis. Given her signs of chronic obstruction and borderline sepsis, urology service opted for a right percutaneous nephrostomy to decompress the right system and prevent urosepsis as well as culture of the upper tract to optimize infectious treatment while pending definitive stone treatment. Her 2.0 cm × 0.9 cm bladder stone, initially projecting into the UVJ on the CT scan four years ago, now appeared to be in the distal right ureter. These findings likely suggest the right ureterocele housing the calculus, causing UVJ obstruction and hydroureteronephrosis with her acute cystitis presentation.

3 DISCUSSION

Ureterocele remains a challenging diagnosis, often eluding detection on routine imaging studies such as CT scans of the abdomen and pelvis. The intricate anatomy of the ureter and its variable presentations contribute to the difficulty in identifying ureteroceles, especially in adult patients. Furthermore, stones within the ureterocele can be misinterpreted as bladder stones on imaging, leading to erroneous diagnoses and persistent symptoms such as pain and recurrent UTIs.

While ureteroceles are well‐recognized in the pediatric population, our understanding in adults relies heavily on case reports and case series[10,11]. The etiology of ureteroceles, characterized by their variable presentations, continues to spark controversy. Two theories are proposed for the formation of ureteroceles: one suggests a congenital origin, while the other implicates an acquired lesion. The most widely accepted theory of ureterocele pathogenesis is a failure of the Chwalla membrane, composed of urogenital sinus tissue and ureteral epithelium, to regress during development. Their variable association along the path of Wolffian duct migration strongly points to more complex abnormal embryogenesis that has yet to be further understood[4,6]. At this time, the acquired argument does not carry as much evidence as the congenital argument[1].

While a number of radiologic modalities may identify ureteroceles, US is usually sufficient to make an initial diagnosis. US typically shows a well‐defined, rounded mass in the posterior bladder that is best visualized with a partially filled bladder[6]. In addition, CT urogram often depicts ureteroceles as a “cobra‐head” deformity where the dilated intravesical portion of the ureter resembles the cobra's head and the extravesical ureter forms the body of the cobra[12]. Cystoscopy may also offer an additional diagnostic and therapeutic option for ureteroceles not clearly elucidated on diagnostic imaging alone[13].

The management of ureteroceles is complex, multimodal, and must be individualized[14]. Options must be tailored to the patient's presentation, age, ureterocele classification, single or duplex collecting system, and presence of infection or stones. Surgical treatment includes resection and endoscopic unroofing with a resectoscope. Most reported cases tend to favor a low transverse incision with Collin's knife, with Holmium and potassium titanyl phosphate lasers recently gaining more traction[3,15,16]. While endoscopic procedures can be used as a minimally invasive option to provide early decompression, they are generally not considered definitive management for patients with ectopic ureteroceles in either a duplex system or presenting with reflux[17–19]. Nevertheless, the goals of ureterocele management prioritize the safeguarding of renal function through relieving obstruction and reflux, halting any bladder wall defects, and eradicating infection and calculi to minimize surgical morbidity.

The potential oversight of ureterocele on imaging underscores the importance of clinical suspicion and thorough evaluation in cases with suggestive symptoms, including flank pain, recurrent UTIs, and hydronephrosis. Healthcare providers should maintain a high index of suspicion for ureterocele, particularly when imaging findings do not correlate with clinical presentation or when there is persistent unexplained urinary symptoms.

In this case, the patient's non‐verbal status secondary to dementia further complicated an already challenging diagnosis. Her initial CT interpretation may have also delayed proper treatment. In cases where there is a discrepancy between symptoms and imaging results, further evaluation with modalities such as US and cystoscopy may be warranted to definitively diagnose ureterocele. Additionally, increased awareness among healthcare providers regarding the deceptive nature of ureterocele presentations can facilitate early recognition and appropriate management, thereby averting potential complications associated with delayed diagnosis and treatment.

4 CONCLUSION

This case describes a 78‐year‐old woman with significant comorbidities who presented with recurrent UTIs, failure to thrive, and an apparently mobile bladder calculus located within the UVJ. Four years later, a CT scan revealed this calculus in the distal right ureter. This case emphasizes not only the wide variability and frequently misleading clinical presentation of ureteroceles but also timely diagnosis to prevent secondary complications of chronic obstruction and renal failure. The potential oversight of ureterocele on imaging studies highlights the need for heightened clinical suspicion and comprehensive evaluation in patients presenting with suggestive symptoms. Failure to recognize ureterocele can lead to misdiagnosis and prolonged morbidity. Healthcare providers must maintain vigilance and consider ureterocele in the differential diagnosis, particularly in cases with recurrent UTIs and persistent urinary symptoms.

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RIGHTS & PERMISSIONS

2024 The Author(s). UroPrecision published by John Wiley & Sons Australia, Ltd on behalf of Higher Education Press.