AUDI Nr. 2_2023

ADVANCES IN UROLOGICAL DIAGNOSIS AND IMAGING EDITOR IN CHIEF Andrea B. Galosi CO-EDITOR Pasquale Martino

OFFICIAL JOURNAL of

S.I.E.U.N.

Italian Society of Integrated Diagnostic in Urology, Andrology, Nephrology

Vol. 6 - n. 2 - 2023

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ATLANTE di ECOGRAFIA UROLOGICA, ANDROLOGICA e NEFROLOGICA a cura di

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ADVANCES IN UROLOGICAL DIAGNOSIS AND IMAGING Official Journal of S.I.E.U.N. EDITOR in CHIEF Andrea B. Galosi, Ancona (IT)

CO-EDITOR Pasquale Martino, Bari (IT)

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Boscutti Giuliano, Trieste (IT), D’Amelio Alessandro, Lecce (IT) Fiorini Fulvio, Rovigo (IT), Gesualdo Loreto, Bari (IT) Granata Antonio, Agrigento (IT), Ranghino Andrea, Ancona (IT)

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Beltran Antonio Lopez, Lisbon (PT), Fiorentino Michelangelo, Bologna (IT) Liang Cheng, Indianapolis (USA), Montironi Rodolfo, Ancona (IT)

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Advances in Urological Diagnosis and Imaging - 2023; 6, 1

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A rare case of giant serous cystadenoma of the spermatic cord Iossa V. , De Luca L. , Napolitano L. , Fusco G.M., Pucci L. , Carrino M.

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Advances in Urological Diagnosis and Imaging - 2023; 6, 1

V. Iossa, L. De Luca, L. Napolitano, G.M. Fusco, L. Pucci, M. Carrino

C ase RepoRt

A rare case of giant serous cystadenoma of the spermatic cord

Iossa V.1, De Luca L.1, Napolitano L.2, Fusco G.M.2, Pucci L.1, Carrino M.1 1 2

Department of Andrology, “Antonio Cardarelli” Hospital, 80131 Naples, (Italy); Department of Urology, “Federico II ” Hospital-Naples, 80131 Naples, (Italy).

Primary tumors of the spermatic cord can be of many types, but serous cystadenoma is particularly rare. We report the clinical case of an 18-year-old patient who presented a large right inguinal swelling that had been progressively increasing in size for approximately 2 years. The mass was painless, mobile, non-reducible upon palpation, and transilluminable. The testicles appeared normal in shape, with no alterations in Doppler flow. An ultrasound confirmed the presence of an inguinoscrotal mass, measuring 6x5 cm, with a multilocular

appearance. A further magnetic resonance imaging (MRI) characterized the mass as a “cystic formation with thin non-vascularized septa.” Subsequently, surgical excision of the neoplasm was performed, preserving the testicle and spermatic cord from which the neoplasm originated. The histological diagnosis revealed a serous cystadenoma, a rare benign tumor that can also originate from the spermatic cord. Clinical suspicion and radiological imaging played pivotal roles in avoiding unnecessary radical surgery.

IntroductIon

case report

Primary tumors of the spermatic cord are rare and can be categorized as either benign (adenomatoid tumor, andiomyofibroblastoma, angioleiomyoma, angiofibrolipoma, dermoid cyst, diverticulum, fibrous pseudotumor, fibrolipoma, mesothelial cyst, myositis ossificans, neuroectodermal tumor, neurofibroma, paraganglioma, spindle cell lipoma) or malignant (adenocarcinoma, angiomyxoma, lymphoma, schwannoma, myxoid liposarcoma, myofibroblastic tumor, osteosarcoma, rhabdomyosarcoma) (tab.1).

An 18-year-old patient, with no significant medical history or previous surgeries, presented with a right inguinoscrotal mass that had been progressively increasing in size over the past 2 years. The mass was painless, mobile, non-reducible upon palpation, and transilluminable (Fig. 1).

SUMMARY

Key words: serous cystadenoma, spermatic cord, cystic tumor.

Fig. 1. The right inguinoscrotal mass.

TAB. 1 Neoplasms of the spermatic cord Benign Neoplasms

Malignant Neoplasms

adenomatoid tumor, angiomyofibroblastoma, angioleiomyoma, angiofibrolipoma, dermoid cyst, diverticulum, fibrous pseudotumor, fibrolipoma, mesothelial cyst, myositis ossificans, neuroectodermal tumor, neurofibroma, paraganglioma, spindle cell lipoma

adenocarcinoma, angiomyxoma, lymphoma, schwannoma, myxoid liposarcoma, myofibroblastic tumor, osteosarcoma, rhabdomyosarcoma

Among these, our group encountered a serous cystadenoma, an adenomatoid tumor, a very rare paratesticular benign neoplasm that originates in the mesothelium. To date, only two other cases have been reported in the literature.

Advances in Urological Diagnosis and Imaging - 2023; 6, 2

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V. Iossa, L. De Luca, L. Napolitano, G.M. Fusco, L. Pucci, M. Carrino

The testicles appeared normal in shape, with no alterations in Doppler flow. Scrotal ultrasound revealed a complex cystic structure measuring a maximum of 6x5 cm with a multilocular appearance in the inguinoscrotal region.

This structure appeared to originate from the posterior aspect of the spermatic cord and showed signs of continuity with the vas deferens at the internal inguinal ring (Fig. 2).

excision were carried out while preserving the vas deferens and other cord elements. The intraoperative histological examination confirmed a serous cystadenoma, a benign neoplasm, thereby avoiding unnecessary radical surgery.

The procedure was completed with anatomical plane closure (Figures 3-4-5). A six-month follow-up examination and ultrasound of the right inguinal region showed no evidence of tumor recurrence.

Fig. 2.

Inguinoscrotal ultrasound of the mass.

Fig. 3. Incision in the right inguinal area, which revealed tight adherence of the mass to the spermatic cord.

To further characterize the mass, a magnetic resonance imaging (MRI) scan was performed, confirming its dimensions and describing it as a “bulky cystic formation containing thin septa, likely originating from the spermatic cord.”

Subsequently, a conservative surgical excision of the parafunicular neoplasm was performed, including an intraoperative histological examination. Following an incision in the right inguinal area, which revealed tight adherence of the mass to the spermatic cord, microdissection and

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Advances in Urological Diagnosis and Imaging - 2023; 6, 2

Fig. 4. Microdissection and excision of the neoplasm

Fig. 5. Macroscopic appearance of the neoplasm.

dIscussIon Serous cystadenoma of the spermatic cord is an exceptionally rare tumor. This is only the fourth reported case since the original report by McCluggage et al. in 1996 (2). The third and last case is reported by Kim et al in 2012 and, among other things, included a case of mucinous cystadenoma of the funiculus, therefore not a serous tumor like the one we treated (3). Moreover, among

V. Iossa, L. De Luca, L. Napolitano, G.M. Fusco, L. Pucci, M. Carrino

these reported cases, our case represents the largest in terms of size, measuring 6x5 cm. Notably, all previously reported patients were over 65 years of age, making it unique that this benign neoplasm affected an 18-yearold patient, as in our case (4). Serous cystadenoma is an epithelial neoplasm that typically exhibits a macroscopic appearance characterized by a smooth, thin, and shiny wall. Microscopically, it consists of a single-layered columnar epithelium with glycogen-rich cells (positive PAS reaction). Often, as in our case, it presents with a multilocular appearance, consisting of small cysts (microcystic variant, “honeycomb” appearance). However, it can also manifest as one or a few larger cysts (oligocystic or macrocystic variant). Serous cystadenoma is a benign neoplasm (5) and is typically asymptomatic, with diagnosis often occurring when its considerable size results in visible aesthetic effects or symptoms due to extrinsic compression of adjacent structures. The macroscopic appearance of the excised mass in our case clearly indicated a funicular origin, as it had completely distorted and displaced the spermatic cord elements, particularly the vas deferens, which was tightly adherent to the cystadenoma at the internal inguinal ring, necessitating careful microdissection to prevent injury. This is why we recommended the patient undergo magnetic resonance imaging (MRI) after the inguinoscrotal ultrasound to assess the anatomical compromise of cord elements before surgical intervention. Following neoplasm excision, despite its macroscopic cystic appearance, we deemed it necessary to perform an intraoperative histological examination to exclude the rare possibility of “borderline” areas or progression to cystadenocarcinoma. This potential outcome has been reported in the pancreas but cannot be definitively ruled out for the spermatic cord due to the limited available data (Serous cystadenomas do not have mutations in either KRAS or BRAF in contrast to serous borderline tumors and serous carcinoma) (6).

dIsclosures Financial Support: No financial support was received for this submission.

Conflict of interest: The authors declare no conflicts of interest.

references 1) Dagur G, Gandhi J, Kapadia K, Inam R, Smith NL, Joshi G, Khan SA. Neoplastic diseases of the spermatic cord: an overview of pathological features, evaluation, and management. Transl Androl Urol 2017;6(1):101-110. doi: 10.21037/tau.2017.01.04 2) McCluggage WG, Shah V, Nott C, Clements B, Wilson B, Hill CM: Cystadenoma of spermatic cord resembling ovarian serous epithelial tumour of low malignant potential: immunohistochemical study suggesting Müllerian differentiation. Histopathology 1996, 28:77–80.

3) Kim JY, Lee YT, Kang HJ, Lee CH. Primary mucinous cystadenoma of the spermatic cord within the inguinal canal. Diagn Pathol 2012; 7: 13 4) Geenen RWF, Bevers RFM, Gielis C, Boon TA: Papillary cystadenoma located in the spermatic cord. J Urol 1997, 158:546.

5) Seidman JD, Mehrotra A. Benign ovarian serous tumors: a re-evaluation and proposed reclassification of serous “cystadenomas” and “cystadenofibromas”. Gynecol Oncol. 2005 Feb;96(2):395-401 6) Strobbel O, Z’Graggen K, Schmitz-Winnenthal FH et al: Risk of malignancy in serous cystic neoplasms of the pancreas. Digestion 2003;68:24-33.

conclusIons Literature provides scarce information regarding serous cystadenoma of the spermatic cord. While undoubtedly a rare neoplasm, it should be considered when ultrasound or MRI reveals the typical characteristics, allowing for a technically feasible surgical excision procedure that preserves the testicle and cord elements. Additionally, due to the limited number of cases in the literature, we recommend performing an intraoperative histological examination because the possibility of “borderline” or cystadenocarcinoma evolution cannot be excluded for the spermatic cord at present. In our case, although the clinical follow-up observation was relatively short (6 months), it is highly likely that conservative surgery was curative (no definite evidence of recurrence during the 6-month follow-up, and no tumor lesions identified outside the inguinal area).

Therefore, recognizing the benign nature of the lesion was essential in avoiding unnecessary radical surgery.

CorrespondenCe Vincenzo Iossa, MD Department of Andrology, “Antonio Cardarelli” Hospital, 80131 Naples, Italy E-mail: vincenzoiossa@msn.com

Advances in Urological Diagnosis and Imaging - 2023; 6, 2

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