
==== Front
Radiol Case Rep
Radiol Case Rep
Radiology Case Reports
1930-0433
Elsevier

S1930-0433(24)00697-6
10.1016/j.radcr.2024.07.111
Case Report
Giant seminal vesicle cystadenoma diagnosed on imagery after acute urinary retention in a middle-aged man: A case report with brief literature review
Barajraji Moncef Al MD Moncef.Al.Barajraji@ulb.be
a⁎
Calderon Victor MD a
Timmermans Luc MD, PhD a
Doerfler Arnaud MD a
Bienfait Lucie MD b
Svistakov Ilyas MD a
a Urology Department, CHU Marie Curie, Charleroi, Belgium
b Histopathology Department, CHU Marie Curie, Charleroi, Belgium
⁎ Corresponding author. Moncef.Al.Barajraji@ulb.be
18 8 2024
11 2024
18 8 2024
19 11 50295032
23 3 2024
18 7 2024
© 2024 The Authors. Published by Elsevier Inc. on behalf of University of Washington.
2024

https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Seminal vesicle cystadenomas (SVC) are exceedingly rare benign tumors, with only 22 cases reported in the literature up to 2018. Here, we present the case of a 40-year-old man who presented with acute urinary retention secondary to a giant pelvic mass. Radiological imaging diagnosed a retrovesical well-delimited multicystic tumor, initially suspected to arise from the left lobe of the prostate but ultimately confirmed to originate from left seminal vesicle. Despite inconclusive biopsy results and normal tumor markers, surgical removal was recommended to confirm diagnosis. The patient initially opted for conservative management but eventually underwent open surgery due to the tumor's significant enlargement on the follow-up imagery. The procedure, though challenging due to the mass's size and proximity to adjacent structures, was successfully completed with minimal blood loss and uncomplicated postoperative phase. Histopathological evaluation confirmed the diagnosis of SVC, marking the largest reported case of its kind and the first to be discovered upon investigation of an acute urinary retention. This case highlights importance of considering SVC in the differential diagnosis of pelvic masses and underscores the role of imagery and surgery together for definitive diagnosis.

Keywords

Seminal vesicle
Cystadenoma
Pelvic MRI
Urology
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pmcIntroduction

Primary tumors originating from seminal vesicles (SV) are exceedingly rare, especially benign tumors [1]. Most SV masses are indicative of malignancies invading SV from adjacent organs, primarily the prostate [1]. Within the category of benign SV tumors, SV cystadenomas (SVC) are especially very uncommon with only 22 cases reported in the literature up to 2018 [2]. While SV tumors can remain asymptomatic and discovered incidentally, they typically manifest with persistent hematuria, hematospermia, or voiding difficulties [[3], [4], [5]]. The differential diagnosis of SV masses requires multimodal approach with clinical assessment (including digital rectal evaluation and tumoral markers study), and especially imagery (transrectal ultrasonography, magnetic resonance imagery [MRI]) while transrectal needle biopsies play a limited role [[2], [3], [4], [5]].

The differential diagnosis is challenging must exclude primary malignant lesions of prostate and rectum as well as inflammatory diseases or abscesses. The preoperative characterization by imagery also helps defining the most appropriated surgical strategy. Indeed, surgical intervention is often indicated for definitive diagnosis and symptoms resolution. Here, we report the case of a 40-year-old man who presented with acute urinary retention after months of dysuria. A computed tomography imagery revealed a giant pelvic mass, with pelvic MRI showing features of cystadenoma of left SV. Preoperative work-up excluded concurrent prostate malignancy before the patient underwent open surgery with retrovesical approach for left vesiculectomy and removal of the cystic mass. The surgery and postoperative stay were uncomplicated, and patient was discharged on third postoperative day. Histological study of the operative specimen confirmed imagery diagnosis of SV cystadenoma. To our knowledge, this is the first case of SVC discovered upon investigation of acute urinary retention and the largest SVC reported yet.

Case description

A 40-year-old man was referred to our department after acute urinary retention 1 week ago. Prior to this event, he had been complaining of dysuria and urgentury for months in a context of lumbar disc herniation, refractory to anticholinergic drugs and antibiotics. His past medical and familial histories were unremarkable, and he had no fertility issues. Catheter withdrawal failed and the patient accepted re-catheterization but refused evaluation by transrectal ultrasonography and digital rectal examination (DRE). Given suspicion of neurogenic bladder, abdominal computed tomography (CT) was ordered to assess upper urinary tract and revealed a giant pelvic multilocular cystic mass (15 × 11 × 16 cm) adjacent to the prostate and displacing bladder and left ureter. The upper urinary tract and other abdominal organs appeared normal, without pathologic pelvic lymph nodes. Abdominal MRI showed a retrovesical well-delimited giant multicystic benign tumor potentially arising from prostate, with respect to adjacent organs. However, transrectal US rather suggested an origin from left seminal vesicle. Transabdominal CT scan-guided biopsy was unconclusive with normal tumor markers (prostate-specific antigen [PSA], carcinoembryonic antigen [CEA], carbohydrate antigen 125 [CA 125]). Surgical removal of the mass was recommended, but the patient refused and favorized intermittent bladder catheterization with close follow-up. A year later, he opted for surgery, and pelvic MRI showed increase in mass size (21 × 21 × 13 cm) without malignant transformation (as shown in Fig. 1). Due to the large size, open surgery with retrovesical approach was decided. Macroscopically, the mass appeared as a large oval and brown tissue with multiple cystic loculations. Due to its size, lateral dissection was challenging, leading to incision and drainage of 2500 mL of a yellow to brownish fluid. Careful dissection allowed to separate the mass from bladder and prostate without injury. The tumor capsule was excised as much as possible with left seminal vesicle. The procedure, though challenging, lasted only 116 minutes with 400mL of blood losses. The postoperative course was uneventful, and he was discharged on third postoperative day. Histopathological study concluded to a left seminal vesicle cystadenoma (as shown in Fig. 2). After 1 month, the patient has regained normal urinary function with normal erectile function. Contrast-enhanced abdomen MRI scan is scheduled at 3 months for the next follow-up visit.Fig. 1 Preoperative abdominal MRI in T2-weighted sequences. A large multicystic pelvic lesion with multiple septations arising from behind the bladder and the pubic symphysis is seen in coronal (A) and sagittal (B) views. The bladder is compressed and displaced laterally but the fat between the tumor and rectum is respected (arrow). The mass is close to the bowel in his superior aspect but remains separated by a fatty tissular layer (arrow head).

Fig 1

Fig. 2 Histopathological analysis of the surgical specimen (H&E, magnification x10 and x100). (A) Microscopically, cut surface of tumor capsule showed multilocular cysts at low magnification (arrow, H&E x10). (B) At higher magnification, the cysts are lined by a single layer of cubic epithelial cells (arrowhead, H&E x100) and the cystic walls are surrounded by a dense fibrous stoma (star, H&E x100) without nuclear or cytoplasmic atypias.

Fig 2

Discussion

Seminal vesicle cystadenomas (SVC) originate from the embryological remnants of Müllerian ducts [1]. Since the initial case reported in 1951 by Soule et al. [6], only 22 cases of SVC have been reported in the literature up to 2018 [2]. According to the most recent review by Dong et al. in 2018, the median age of patients diagnosed with SVC was 48 years (range 23-71 years) with a median tumor diameter of 8.8 cm (range 3-17.2 cm) [2]. The largest SVC reported in this review measured 17.2 cm in diameter in a 45-year-old man experiencing dysuria and intermittent pelvic pain [7]. As in our case, SVC seems more prevalent in middle-aged men. Clinically, SVC often present with variable symptoms such as hematuria, dysuria, hemospermia, abdominal pain, or painful defecation [[2], [3], [4]]. Sometimes, reduced ejaculate volume or infertility may even be the only feature [5]. This variability in clinical expression likely comes from differences in the mass size and location [4]. Interestingly, examples of SVC discovered after acute urinary retention have not been reported yet. When dealing with an SV mass, the definitive diagnosis relies mainly on surgery to allow histological evaluation, since preoperative needle aspiration/biopsy is often unconclusive [1,2,8]. Nonetheless, imagery plays a crucial role preoperatively in detecting and characterizing the tumor, especially pelvic MRI which allows to establish the origin and nature of the mass, and its precise anatomical delineation for surgical planning [5]. Pelvic MRI often shows a multiseptate and well-delineated encapsulated cystic mass originating from the level of seminal vesicles and located between the bladder and rectum [[2], [3], [4]]. It is important to note that diagnostic workup should always exclude congenital abnormalities of upper urinary tract, especially renal agenesis [5]. Preoperatively, evaluation of tumoral markers such as PSA, CEA and CA 125 may help differential diagnosis with SV carcinoma (CA125), prostate cancer (PSA), or intestinal malignancies (CEA), the 3 of which are normal in SVC [2]. Histologically, SVC present as a well-circumscribed multilobulated mass containing yellow to brownish fluid [[1], [2], [3]]. Microscopically, SVC is characterized by a single columnar or cuboid epithelial cells layer lining cystic areas filled with homogenous eosinophilic material and surrounded by a fibrous stroma [[1], [2], [3]]. Immunohistochemistry staining of SVC shows epithelial positivity for cytokeratin and negativity for calretinin and PSA, with no reactivity of stromal cells for S-100 protein [3]. A rare histologic subtype of SVC with an epithelial papillary component has also been reported [9]. As of now, there are no established recommendations for treatment of SVC. Surgery is often indicated for symptomatic cases or asymptomatic cases independently from tumor size, as cystic SV tumor may be malignant and larger tumors are more challenging to excise with increased risk of injuring adjacent structures [[2], [3], [4], [5],[8], [9], [10]]. Most published reports describe open surgeries with various approaches (transperineal, transvesical, paravesical, retrovesical) [8]. However, despite its excellent results, open surgery is more difficult due to the deep position of SV in pelvis with increased risk of injury to rectum and bladder, ureters, and neurovascular structures) [5,8]. Consequently, laparoscopic approach of vesiculectomy has been increasingly reported nowadays [[2], [3], [4]] as well as robotic-assisted transperitoneal procedures [5,7,10]. Both laparoscopic/robotic or open surgery have excellent outcomes, but the choice also depends on the mass location and size [[2], [3], [4]]. In our case, open surgery with retrovesical approach was the only viable option because of tumor size and location. As of now, the best follow-up strategy after SVC removal has not been established yet. Local recurrences are rare and occurred in only 2 of the 22 patients (9,1%) in the review by Dong et al, after 2 years and 3 years [2]. Incomplete tumoral excision may increase risk of recurrence [4]. Therefore, negative surgical margin should always be obtained if feasible, using intraoperative frozen section if needed [10].

Conclusion

Primary tumors of SV are very rare, especially cystadenomas. SV cystadenomas have variable clinical expression, but acute urinary retention as initial manifestation has never been reported yet. We herein report the largest case of SV cystadenomas yet in a 40-year-old man, and the first to present initially with acute urinary retention. While imagery plays an important role in initial diagnostic work-up of SV masses, surgery is often indicated to resolve symptoms and confirm diagnostic. During surgery, care should be taken to avoid adjacent organ injuries with complete tumoral excision to avoid recurrence. Compared to open surgery, laparoscopic or robotic-assisted surgery offers lower morbidity with similar oncological outcomes.

Consent for publication

Approval for publication was granted from our institution.

Patient consent

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

Competing Interests: The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.
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