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  Table of Contents    
CASE REPORT  
Year : 2016  |  Volume : 59  |  Issue : 3  |  Page : 372-375
Sertoli cell tumor arising in a cryptorchid testis presenting as a content of inguinal hernial sac


1 Department of Pathology, Kempegowda Institute of Medical Sciences, Bengaluru, Karnataka, India
2 Department of Surgery, Kempegowda Institute of Medical Sciences, Bengaluru, Karnataka, India

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Date of Web Publication10-Aug-2016
 

   Abstract 

Sertoli cell tumors (SCTs) are rare tumors accounting for <1% of all testicular tumors. Here, we report a rare case of SCT in a 60-year-old man presenting as a painless swelling in the right groin since childhood. Clinically, he presented with right-sided inguinal hernia with absence of the right testis. He had normal left testis and had no gynecomastia or infertility. The specimen of hernial sac showed testis with a 1.6 cm × 1.5 cm nodular mass having gray tan-cut surface. Histopathologically, the testis showed atrophy and the nodular portion showed tumor cells arranged in tubular and microcystic pattern, with no solid pattern or necrosis. The diagnosis of SCT was confirmed with immunohistochemical staining for inhibin which showed fine granular cytoplasmic positivity. Cryptorchid testis having SCT and presenting as a content of inguinal hernia is a rare occurrence.

Keywords: Cryptorchid testis, hernial sac, Sertoli cell tumor

How to cite this article:
Venkatesh K, Hemalata M, Sathyavathi S, Kumar S. Sertoli cell tumor arising in a cryptorchid testis presenting as a content of inguinal hernial sac. Indian J Pathol Microbiol 2016;59:372-5

How to cite this URL:
Venkatesh K, Hemalata M, Sathyavathi S, Kumar S. Sertoli cell tumor arising in a cryptorchid testis presenting as a content of inguinal hernial sac. Indian J Pathol Microbiol [serial online] 2016 [cited 2019 Oct 23];59:372-5. Available from: http://www.ijpmonline.org/text.asp?2016/59/3/372/188135



   Introduction Top


In a cryptorchid testis, germ cell tumors are common and non-germ cell tumors such as Sertoli cell tumor (SCT) are very rare. SCTs are part of a group of sex cord stromal tumors together with Leydig cell, granulosa cell, and mixed tumors [1] and are considered uncommon because they account for only 1% of all cases diagnosed.[2],[3] Majority of the SCTs are found between 15 and 80 years of age with a mean age of 45 years. The WHO classification includes SCTs and the three variants, namely, sclerosing SCT, lipid-rich SCT, and large cell calcifying SCT.[4] Risk of SCT is found to be higher in patients with cryptorchid testis than in a normally descended testis.[5],[6]


   Case Report Top


A 60-year-old male patient presented with a history of swelling in the right groin since childhood with recent increase in size. Clinical examination revealed absence of the right testis in the scrotal sac. The contralateral testis was unremarkable. Contrary to the literature, the patient had no history of infertility or gynecomastia.

A clinical diagnosis of the right inguinal hernia with undescended testis was made, for which Kuntz procedure with hernioplasty was performed.

Pathological findings

Grossly, hernial sac with the right testis measuring 23.0 cm × 10.0 cm × 0.5 cm was received. Testis measured 4.5 cm × 2.0 cm × 1.0 cm. Cut section of the testis showed positive string sign and a well-circumscribed nodule measuring 1.6 cm × 1.5 cm × 1.0 cm. Cut section of the nodule was gray tan with many tiny cystic spaces [Figure 1] and [Figure 2].
Figure 1: Gross: Hernial sac and testis with attached well-circumscribed nodule

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Figure 2: Close-up view of the testis with the gray tan nodule (gross)

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Microscopic examination from the nodule revealed a well-circumscribed tumor with cells arranged in tubules, sheets, and microcystic pattern. Individual cells were oval to columnar with abundant vacuolated granular pale eosinophilic cytoplasm and vesicular nuclei. Cells exhibited mild pleomorphism and infrequent mitosis. Stroma was less cellular and hyalinized at places [Figure 3] and [Figure 4].
Figure 3: Neoplasm with adjacent testis showing hyalinized tubules (H and E, ×100)

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Figure 4: Neoplastic cells in tubular pattern and intervening sclerosed stroma (H and E, ×100)

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Rest of the testis showed hyalinized atrophic tubules without germ cells and Leydig cell hyperplasia in the interstitium. Sections from the hernial sac showed fibrocollagenous wall with congested blood vessels and focal collection of lymphocytes.

Based on the above features, a diagnosis of SCT arising in a cryptorchid atrophic testis presenting as a content of hernial sac was made.

Immunohistochemical staining for inhibin showed fine granular cytoplasmic positivity which further confirmed the diagnosis [Figure 5] and [Figure 6]. Tumor cells were weakly positive for cytokeratin and vimentin and were negative for placental alkaline phosphatase (PLAP).
Figure 5: Tumor cells showing positivity for inhibin (H and E, ×100)

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Figure 6: Tumor cells showing fine granular cytoplasmic positivity for inhibin (IHC, ×400)

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   Discussion Top


Testicular tumors are relatively rare with approximately 95% of tumors being of germ cell origin while the rest being sex cord stromal tumors.[2],[4] Among sex cord stromal tumors, SCTs are rarer constituting <1%, and half of these tumors (<0.5%) arise in a cryptorchid testis with peak incidence at 35–50 years of age.[2],[7] In total, only 170 cases have been reported in literature.[3] The first sign is usually a painless mass [8] and the serum levels of alpha-fetoprotein, beta-human chorionic gonadotropin, and lactate dehydrogenase are within the normal limits. As for all testicular tumors, the risk of developing a SCT increases in patients with undescended testicles.[3] SCTs are observed as nodular masses that are easily distinguished from testicular tissue during ultrasound examination. Patients present with a scrotal mass which is usually unilateral, but bilateral cases have been reported. Approximately, 20% of the patients have gynecomastia, more commonly associated with malignant forms, and the tumor can be seen in patients with undescended testes,  Peutz-Jeghers syndrome More Details, and testicular feminization syndrome.[6]

The classical SCT is most common, and has a highly variable range of pathological characteristics. It has a tubular pattern, with cells having pale or eosinophilic cytoplasm, vesicular nucleus, and hyalinized stroma.[1] In complicated cases, immunohistochemistry is performed which shows positivity for cytokeratin and inhibin, and it is negative for PLAP.[9]

The SCT of large cell calcifying type is another variant occurring at an earlier age. It mainly affects children and young adults aged approximately 20 years, and shows a solid growth pattern, polygonal cells, prominent nucleolus, and calcifications.[8],[9]

The sclerosing variant is considered extremely uncommon, with only 14 cases being reported till date.[10] This tumor shows small tubules in a sclerotic stroma, and it is usually negative or weakly positive for cytokeratin.[9]

Sex cord tumors such as SCTs form well-defined tubular structures.[11] SCT of the testis is a rare sex cord stromal tumor composed of cells expressing varying degrees of features of fetal, prepubertal, or adult  Sertoli cells More Details. Malignant transformation occurs in nearly 10% of the testicular tumors. The incidence of testicular tumors is higher in a cryptorchid testis [2] than in a normally descended testis and rarely may present as a content of hernial sac. The surgeon should always be alert to the possibility of cryptorchid testis during a surgical exploration for inguinal hernia.[5] SCT stains positive for inhibin, vimentin, and cytokeratin.[12],[13]

SCT should be distinguished from small Sertoli cell nodules, Sertoli cell adenoma, rete testis adenoma, and Leydig cell tumors.[14]

Considering the higher risk of development of SCTs in both the gonads in a patient with unilateral cryptorchid testis, long-term follow-up is recommended.


   Conclusion Top


According to the available literature, SCT is a rare tumor arising in a cryptorchid testis and its presentation as a content of hernial sac is almost a nonentity. Based on the histopathological features, diagnosis of SCT in a cryptorchid testis was made which was confirmed by immunohistochemistry in this case. We conclude that a diagnosis of non-germ cell tumors such as SCT should be considered in a patient presenting with cryptorchid testis, particularly in elderly although common tumors are from the germ cells. A routine histopathological study along with immunohistochemistry to complement it is necessary in making the correct diagnosis.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Young RH. Sex-cord tumors of the testis. Pathol Case Rev 2005;10:193-205.  Back to cited text no. 1
    
2.
Young RH, Koelliker DD, Scully RE. Sertoli cell tumors of the testis, not otherwise specified: A clinicopathologic analysis of 60 cases. Am J Surg Pathol 1998;22:709-21.  Back to cited text no. 2
    
3.
Ozturk H, Saracoglu M, Zengin T, Sivrikoz ON, Kerman HS, Adakan S. Asymptomatic Sertoli cell tumour diagnosed during azoospermia work-up. Asian J Androl 2013;15:845-6.  Back to cited text no. 3
    
4.
Anderson GA. Sclerosing Sertoli cell tumor of the testis: A distinct histological subtype. J Urol 1995;154:1756-8.  Back to cited text no. 4
    
5.
Kassir R, Dubois J, Berremila SA, Baccot S, Boueil-Bourlier A, Tiffet O. A rare variant of inguinal hernia: Cryptorchid testis at the age of 50 years. Etiopathogenicity, prognosis and management. Int J Surg Case Rep 2014;5:416-8.  Back to cited text no. 5
    
6.
Eble JN, Sauter G, Ebstein JI, Sesterhenn IA, editors. World Health Organization Classification of Tumours. Pathology and Genetics of Tumours of the Urinary System and Male Genital Organs. Lyon: IARC Press; 2004. p. 217-78.  Back to cited text no. 6
    
7.
Giglio M, Medica M, De Rose AF, Germinale F, Ravetti JL, Carmignani G. Testicular Sertoli cell tumours and relative sub-types. Analysis of clinical and prognostic features. Urol Int 2003;70:205-10.  Back to cited text no. 7
    
8.
Gómez García I, Romero Molina M, López-García Moreno A, Buendía González E, Rubio Hidalgo E, Bolufer E, et al. Sertoli cell tumor, a rare testicular tumor, our experience and review of the literature. Arch Esp Urol 2010;63:392-5.  Back to cited text no. 8
    
9.
Jiménez JD, Cebrián JL, Guarch R, Hualde A. Sertoli cell tumor of the testis with positive neuroendocrine markers. Actas Urol Esp 2010;34:481-3.  Back to cited text no. 9
    
10.
Werther M, Schmelz HU, Schwerer M, Sparwasser C. Sclerosing Sertoli cell tumor of the testis: A rare tumor. Case report and review of the literature on the subtypes of Sertoli-cell tumor. Urologe A 2007;46:1551-6.  Back to cited text no. 10
    
11.
Zukerberg LR, Young RH, Scully RE. Sclerosing Sertoli cell tumor of the testis. A report of 10 cases. Am J Surg Pathol 1991;15:829-34.  Back to cited text no. 11
    
12.
Govender D, Sing Y, Chetty R. Sertoli cell nodules in the undescended testis: A histochemical, immunohistochemical, and ultrastructural study of hyaline deposits. J Clin Pathol 2004;57:802-6.  Back to cited text no. 12
    
13.
Iczkowski KA, Bostwick DG, Roche PC, Cheville JC. Inhibin A is a sensitive and specific marker for testicular sex cord-stromal tumors. Mod Pathol 1998;11:774-9.  Back to cited text no. 13
    
14.
Barghorn A, Alioth HR, Hailemariam S, Bannwart F, Ulbright TM. Giant Sertoli cell nodule of the testis: Distinction from other Sertoli cell lesions. J Clin Pathol 2006;59:1223-5.  Back to cited text no. 14
    

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Correspondence Address:
Dr. S Sathyavathi
59/2, 31st C Cross, Corporation Layout, 4th T Block, Jayanagar, Bengaluru - 560 041, Karnataka
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0377-4929.188135

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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