Unusual presentation of cowper gland adenocarcinoma Mimicking post-traumatic urethral stricture

Abstract

Urethral strictures are commonly attributed to trauma, inflammation, or iatrogenic causes, but malignancies are a rare etiology. We report a case of a 57-year-old male with a history of trauma presenting with a bulbomembranous urethral stricture, initially suspected as post-traumatic. Intraoperative frozen section analysis unexpectedly revealed Cowper gland adenocarcinoma, leading to definitive surgical management with radical cystoprostatectomy and urethrectomy. This case underscores the importance of considering malignancy in atypical stricture presentations to ensure accurate diagnosis and appropriate treatment.

Highlights

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Cowper gland adenocarcinoma is a rare but critical differential in urethral strictures.
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Malignancy should be considered in atypical urethral stricture presentations.
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Intraoperative frozen section analysis aided in the unexpected urethral cancer diagnosis.
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Radical cystoprostatectomy and urethrectomy were necessary for definitive treatment.
•Early recognition of Cowper malignancy can prevent delays in appropriate management.

1 Introduction

Urethral strictures are characterized by the narrowing of the urethra, which leads to obstructed urinary flow. The etiology of anterior urethral stricture disease may be broadly subcategorized into iatrogenic, traumatic, inflammatory, and idiopathic causes. However, malignancies, though rare, can also lead to urethral strictures. This case report discusses an unusual presentation of urethral malignancy in a patient with a history of trauma who was found to have obliteration of the bulbomembranous urethra, a classic radiologic appearance seen with pelvic fracture urethral injury (PFUI). The report emphasizes the importance of thorough history taking and complete diagnostic evaluation when contemplating posterior urethroplasty. By detailing this case, we hope to contribute to the understanding and recognition of rare causes of urethral strictures, ultimately improving patient outcomes through timely and accurate diagnosis.

2 Case report

A 57-year-old Vietnamese male sustained a work-related fall from a height of 3 m onto concrete, and subsequently developing urinary retention. He presented to an Emergency Department where attempts to place a catheter were unsuccessful, leading to the placement of a suprapubic tube. He was referred to a tertiary referral center for urethral stricture disease in Ho Chi Minh City, Vietnam. One month after suprapubic tube placement, a diagnostic evaluation including antegrade cystoscopy, retrograde urethrogram, and cystourethrogram revealed a 4–5 cm bulbomembranous posterior urethral obliterative stricture ( Fig. 1 ). The patient was scheduled for posterior urethroplasty during an annual Urethroplasty Workshop with Visiting Professors.

Prior to surgery, the patient was interviewed to clarify details of his history. He reported that he did not seek medical attention at the time of the trauma due to a lack of symptoms. He then developed rapidly progressive obstructive voiding symptoms approximately 6-months after the injury leading to subsequent retention. The patient had no history of smoking, recreational drug use, or significant past medical history. A physical examination revealed normal genitourinary findings.

Upon reviewing the patient’s history and imaging results, concerns were raised about the potential underlying cause because in general, when there is trauma associated with a long urethral defect, the patient is in retention immediately after the injury. Moreover, when there is a fall, this can lead to a straddle trauma, which typically involves the proximal bulbar urethra or PFUI where the urethra is typically obliterated after urethral rest with extension into the membranous urethra proximally, which was the case for this patient. However, the patient did not suffer a pelvic fracture at the time of his trauma. Due to the unusual presentation and severity of the stricture, the pathology department was alerted in advance for the possibility of requiring a frozen section to rule out any unknown etiologies, and possible biopsy of the urethra was included in the surgical consent.

The patient was anesthetized and placed in a high lithotomy position. A lambda incision was made in the perineum to expose the urethra. It was observed that the proximal bulbous urethra tissue was firm and abnormally stiff, which prompted the decision to send a sample of the urethral tissue for frozen section analysis. The frozen section pathology results revealed adenocarcinoma. Given these unexpected findings, additional urethral tissue samples were taken for detailed postoperative pathology, and we did not proceed with excision and primary anastomosis. The patient’s surgical site was then closed, and he was discharged with a suprapubic catheter for further management.

The permanent pathology results confirmed the diagnosis of Cowper gland adenocarcinoma of the urethra. Immunohistochemical staining showed CK7 (+), PSA (−), GATA 3 (−), CDX 2 (−), CK5/6 (+), which supported the diagnosis. Following these findings, the patient was scheduled for further surgical intervention to address the malignancy.

Unfortunately, the patient was lost to follow-up against medical advice and returned six months later. An MRI scan showed significant abnormalities in the proximal corpus spongiosum and prostatic urethra that extended to the bladder neck and surrounding the prostate, with involvement of the levator ani muscle and urethral sphincter ( Fig. 2 ). The metastatic workup was negative, and colonoscopy revealed no rectal involvement. Given the MRI findings and lack of metastatic disease, the patient underwent a radical cystoprostatectomy with urethrectomy.