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  Table of Contents    
CASE REPORT  
Year : 2022  |  Volume : 65  |  Issue : 2  |  Page : 440-443
An unusual testicular mass in a young male: Metastasis from occult gastric carcinoma


1 Department of Pathology, All India Institute of Medical Sciences, New Delhi, India
2 Department of Medical Oncology, All India Institute of Medical Sciences, New Delhi, India

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Date of Submission13-Aug-2020
Date of Decision24-Mar-2021
Date of Acceptance04-Oct-2021
Date of Web Publication14-Apr-2022
 

   Abstract 


Metastases to the testis are uncommon. Signet-ring cell carcinomas from the gastrointestinal tract (GIT) can rarely disseminate to the testicles, mimicking primary testicular malignancies with signet-ring cells. We hereby describe a case of a 26-year-old male who presented with left testicular swelling, multiple lymphadenopathies, and normal serum tumor markers. Lymph node biopsy revealed clusters and singly lying signet-ring cells. Judicious use of immunohistochemistry confirmed the tumor to be GIT primary. Further investigations confirmed a gastric tumor extending to the duodenum. Although rare, metastatic tumors to the testis should be considered in differential diagnoses of testicular masses in a young patient, particularly when serum germ cell tumor markers are normal or mildly deranged.

Keywords: Gastrointestinal tract, immunohistochemistry, metastasis, signet-ring cell carcinoma, testicular tumor

How to cite this article:
Nakra T, Sharma A, Sharma A, Kakkar A. An unusual testicular mass in a young male: Metastasis from occult gastric carcinoma. Indian J Pathol Microbiol 2022;65:440-3

How to cite this URL:
Nakra T, Sharma A, Sharma A, Kakkar A. An unusual testicular mass in a young male: Metastasis from occult gastric carcinoma. Indian J Pathol Microbiol [serial online] 2022 [cited 2023 Feb 5];65:440-3. Available from: https://www.ijpmonline.org/text.asp?2022/65/2/440/343216





   Introduction Top


Metastases to the testes are rare, accounting for 0.02%–2.5% of testicular tumors, and usually affect older adults.[1],[2] The commonest primary site of origin of metastases in adults is the prostate, followed by the gastrointestinal tract (GIT), lung, kidney, and urinary bladder.[1],[2] Metastases occur either synchronously or metachronously with the primary neoplasm. Unlike its ovarian analog, Krukenberg tumor, testicular metastases from the GIT are infrequent, with the stomach being the commonest primary site.[2] Criteria that favor a testicular tumor being metastatic include older patient age, bilateral involvement, known extra-testicular malignancy, uncommon morphology for a primary testicular tumor, intertubular infiltrative pattern, and lymphovascular emboli.[2] As histologically similar tumors involving the GIT and testis could have either site as primary, immunohistochemistry can be helpful in arriving at the correct diagnosis, necessary for appropriate management.

We report the case of a young adult who presented with testicular swelling and cervical lymphadenopathy, clinically mimicking a primary testicular tumor with lymph node metastases. Histopathology supported by immunohistochemistry revealed it to be a metastatic carcinoma; endoscopic evaluation led to the identification of a primary gastric carcinoma confirmed on biopsy.


   Case Report Top


This 26-year-old male presented with a left-sided neck swelling for 3 months, along with decreased appetite, weight loss, and multiple bone pains. Physical examination revealed enlarged left cervical, axillary, and bilateral inguinal lymph nodes, hard in consistency. There was also a firm to hard left testicular swelling. CT neck, chest, and abdomen showed necrotic lymph nodes in the posterior triangle of the neck, right iliac fossa, peripancreatic region, and mediastinum. Serology revealed elevated lactate dehydrogenase (LDH) (1563 U/L), while β-human chorionic gonadotropin (HCG) (24.5 mIU/mL) alpha-fetoprotein (AFP) were within normal limits (0.5 ng/mL). The possibility of a metastatic testicular tumor was considered, and a cervical lymph node excision biopsy was performed.

Microscopy showed a tumor infiltrating the subcapsular sinuses, and at places replacing the lymph nodal tissue. Tumor cells were discohesive, present in large clusters and as singly infiltrating cells with signet-ring cell morphology, having an eccentric nucleus and intracytoplasmic mucin highlighted by Alcian blue [Figure 1]a, [Figure 1]b, [Figure 1]c, [Figure 1]d. Morphological features of germ cell tumors (GCT) were not identified. Immunohistochemistry revealed diffuse positivity for pancytokeratin, CK7 and CK20, with focal weak positivity for CDX2; SALL4, PLAP, and CD117 were negative, excluding a GCT [Figure 1]e, [Figure 1]f, [Figure 1]g, [Figure 1]h, [Figure 1]i. A diagnosis of metastatic signet-ring cell carcinoma was offered, with a possible primary in the GIT. Upper gastrointestinal endoscopy revealed unhealthy, thickened gastric mucosa extending to the first part of the duodenum. A biopsy showed similar tumor cells infiltrating the lamina propria of the duodenal mucosa [Figure 2]. A final diagnosis of primary gastric poorly cohesive carcinoma with signet-ring cells metastatic to the testis and lymph nodes was made. The patient received one cycle of 5-fluorouracil-based palliative chemotherapy, after which he was lost to follow-up.
Figure 1: Photomicrographs of excised lymph node showing infiltration by tumor cells (a) in clusters as well as lying singly (b). The tumor cells have signet-ring–like morphology (c), and contain acidic mucin (d; Alcian Blue-Periodic acid Schiff). Tumor cells are diffusely immunopositive for CK7 (e), CK20 (f), and focally for CDX2 (g) while they are negative for SALL4 (h) and CD117 (i). [a-c, H&E; e-i, IHC]

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Figure 2: Photomicrographs of duodenal biopsy show groups of signet-ring–like cells (arrow) in the lamina propria (a, b; H&E). These tumor cells contain acidic mucin on Alcian Blue- Periodic acid Schiff stain (c). Immunohistochemically, the tumor cells are negative for CDX2 unlike normal duodenum (d; IHC)

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   Discussion Top


Metastases to the testes from solid tumors are rare, with prostatic adenocarcinoma being the commonest. Possible pathways of tumor dissemination include hematogenous route, retrograde through lymphatics, retrograde extension along the spermatic cord, and seeding of peritoneal surfaces including tunica vaginalis.[1] However, the relatively low temperature of the scrotum makes the environment unfavorable for the survival of metastatic tumor cells, explaining the rarity of testicular metastases.[3]

Metastases to testis from GIT, and vice versa, are both reported in the literature.[4] In the GIT, the stomach is the commonest primary site; metastases from the caecum and appendix are also described.[1],[5] Less than 20 cases of gastric carcinoma with testicular metastasis are documented, four of which had initial presentation as a scrotal swelling.[6],[7],[8],[9] The majority of these presented in the 5th to 8th decades of life, with only one patient presenting in his twenties, like the present case.[9]

As management depends upon the primary diagnosis, it is imperative to distinguish metastases from a primary testicular neoplasm. This distinction cannot be made clinically or radiologically, unless there is a previous history of extra-testicular malignancy. In around 10% of patients, testicular enlargement may be the first presentation of a malignancy elsewhere,[2] as in our case. However, unlike the present case, secondary tumors usually occur in the 5th to 7th decades of life, whereas primary tumors are rare over 40 years of age. Clinical presentation is similar in both conditions, with a painless increase in the volume of the testicle. Unlike metastases to ovaries, most metastases to the testes are unilateral; bilateral involvement occurs in only 15%–20% of cases.[2] Elevated serum tumor markers for GCTs like AFP, β-HCG, and LDH favor a testicular origin, whereas normal tumor markers should raise the possibility of metastasis, as observed in this case. Conversely, elevated CA 19-9 and carcinoembryonic antigen favor metastasis over a primary.

Although a variety of tumors from different sites show signet-ring cell morphology, it is usually appreciated focally in the background of classical tumor morphology. The presence of solely signet-ring cells at metastatic sites raises suspicion of the GIT as the possible primary site. However, in a young patient presenting with a testicular mass, the differential diagnosis of primary testicular tumors with signet-ring cells does arise. GCTs including seminoma and yolk sac tumor can show signet-ring cell features; however, classical features of the GCT would be evident at least focally.[2] Furthermore, the presence of germ cell neoplasia in situ (GCNIS) in the vicinity of a testicular neoplasm would favor a primary testicular GCT over metastasis. Immunohistochemistry can aid in determining cell lineage and tumor origin [Table 1]. The most commonly used cytokeratins, CK7 and CK20, can be used to characterize and differentiate between signet-ring cell carcinomas of the stomach, colon, and breast.[10] Rarely, gastric carcinoma may express AFP or β-HCG, causing difficulty in making the distinction.[1] In our case, diffuse cytokeratin positivity with negative GCT markers favored a primary GIT tumor, metastasizing to the testis over a metastatic GCT and was further supported by endoscopic findings. Lastly, the remote possibility of a second primary tumor in the testis also needs to be excluded. Considering the young age of our patient, the most likely primary testicular malignancy would be a GCT. But the incidence of two synchronous primaries with near-normal serum tumor markers is highly unlikely; thus, the possibility of testicular metastasis was favored in this case.
Table 1: Histochemical and immunohistochemical stains for differential diagnosis of testicular tumors

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   Conclusion Top


In a young male with a testicular mass, metastatic malignancy should be considered, particularly in the setting of normal serum GCT markers. Relevant investigations to identify a primary, including a biopsy should be obtained. Histopathological examination and appropriate immunohistochemical work-up can reliably identify the site of the primary tumor, which is required for planning therapy.

Highlights

  • Metastasis to the testis should be considered in differential diagnoses of a testicular mass in a young patient, especially in the setting of normal serum germ cell tumor markers.
  • Testicular metastasis of signet-ring cell carcinomas from the gastrointestinal tract is rare and can mimic primary testicular malignancies.
  • Histopathological examination with the judicious use of immunohistochemistry can reliably identify the primary site of the tumor.


Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
   References Top

1.
Li B, Cai H, Kang ZC, Wu H, Hou JG, Ma LY. Testicular metastasis from gastric carcinoma: A case report. World J Gastroentero 2015;21:6764-8.  Back to cited text no. 1
    
2.
Gonzalez-Peramato P, Nistal M, Ulbright TM. Metastatic tumors. In: Moch H, Humphrey PA, Ulbright TM, Reuter VE, editors. WHO Classification of Tumors of the Urinary System and Male Genital Organs. 4th ed. Lyon: IARC; 2016. p. 257-8.  Back to cited text no. 2
    
3.
Mazumdar S, Sundaram S, Patil P, Mehta S, Ramadwar M. A rare case of metastatic germ cell tumor to stomach and duodenum masquerading as signet ring cell adenocarcinoma. Ann Transl Med 2016;4:309.  Back to cited text no. 3
    
4.
Bodon GR, Dressler JA. Metastatic carcinoma of right testicle from primary carcinoma of the appendix. J Urol 1967;97:885-7.  Back to cited text no. 4
    
5.
Muir GH, Fisher C. Gastric carcinoma presenting with testicular metastasis. Br J Urol 1994;73:713-4.  Back to cited text no. 5
    
6.
Kollas A, Zarkavelis G, Goussia A, Kafantari A, Batistatou A, Evangelou Z, et al. Testicular signet-ring cell metastasis from a carcinoma of unknown primary site: A case report and literature review. Case Rep Oncol Med 2016; 2016:Article ID 7010173, 5 pages. https://doi.org/10.1155/2016/7010173.  Back to cited text no. 6
    
7.
Ozdal OL, Yakupoglu YK, Cicek A, Erdem O, Memis L, Memis A. Epididymal metastasis from gastric signet ring cell adenocarcinoma. Can J Urol 2002;9:1498-9.  Back to cited text no. 7
    
8.
Schaefer IM, Sauer U, Liwocha M, Schorn H, Loertzer H, Füzesi L. Occult gastric signet ring cell carcinoma presenting as spermatic cord and testicular metastases: “Krukenberg tumor” in a male patient. Pathol Res Pract 2010;206:519-21.  Back to cited text no. 8
    
9.
Yang KC, Chao Y, Luo JC, Kuo JY, Lee RC, Li AF, et al. The unusual presentation of gastric adenocarcinoma as a testicular mass: A favorable response to docetaxel and Cisplatin plus oral tegafur/uracil and leucovorin. J Chin Med Assoc 2010;73:88-92.  Back to cited text no. 9
    
10.
Jagtap SV, Nikumbh DB, Khirsagar AY, Jagtap SS, Shamima. Primary signet-ring carcinoma (Linitus Plastica) of the colorectum presenting as subacute intestinal obstruction. Online J Health Allied Scs 2012;11:14.  Back to cited text no. 10
    

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Correspondence Address:
Aanchal Kakkar
Department of Pathology, All India Institute of Medical Sciences, Ansari Nagar, New Delhi - 110 029
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/IJPM.IJPM_976_20

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