Case Presentation: 57-year-old male patient with past medical history of major depression, essential hypertension and coronary artery disease status post drug-eluting stent placement who presented to the Genitourinary Medical Oncology Clinic due to marked swelling of his left scrotum. Ultrasound showed a hypoechoic solid mass with large left hydrocele. Tumor markers were negative. Computed tomography (CT) of the chest, abdomen and pelvis demonstrated enlarged para-aortic lymph nodes. He underwent an inguinal orchiectomy that demonstrated keratinizing squamous cell carcinoma (SCC). All surgical margins were negative and germ cell neoplasia in-situ was not identified. Positron Emission Tomography/CT showed retroperitoneal metastasis. He underwent surgical resection that included ureterolysis, cord excision, mobilization and resection of a portion of the descending colonic mesentery and serosa with lysis of adhesions. Three months later, surveillance imaging revealed progression of metastatic disease including a mass between the transverse duodenum and inferior vena cava invading the duodenal wall without obstruction. Two days later, he presented to the hospital due to gastrointestinal bleeding. CT of the abdomen was negative for a retroperitoneal bleed or intraluminal bleed with stable metastatic retroperitoneal lymph nodes. Esophagogastroduodenoscopy showed a 2-cm fungating and oozing mass in the second portion of the duodenum. A biopsy from the small bowel mass confirmed metastatic keratinizing SCC. Palliative radiation was initiated in an attempt to control bleeding.

Discussion: A painless testicular mass is the most common initial presentation of testicular cancer. Germ cell tumors (GCTs) account for more than 90% of testicular cancer. Primary testicular squamous cell carcinoma (tSCC) is extremely rare, and metastasis from other organs should be excluded first. Lymphoma and prostate cancer are the most common disease to metastasize to the testicles. Among SCC, lung is the most common primary site. To date, only five cases of tSCC have been reported in the literature. Our patient is similar to the cases reported in the literature in terms of age, longstanding history of testicular enlargement with rapid growth and negative tumor markers. Similar to other cases, our patient, perhaps, had epidermal cyst or hydrocele for a long time which transformed into SCC leading to rapid growth. In contrast, our patient had extensive tumor burden due to lymphovascular invasion and underwent excision of tumor and retroperitoneal lymph node dissection with a curative intent. However, the tumor had rapid metastatic progression after three months leading to intestinal bleeding requiring palliative radiation on two separate occasions and ongoing systemic chemotherapy. In addition, gastrointestinal involvement from testicular cancer is very uncommon (5%) with only 1.4% of cases demonstrating duodenal metastases. To date, this represents the first case of tSCC with gastrointestinal involvement reported in the literature.

Conclusions: Squamous cell carcinoma of the testicles, though rare, is an aggressive malignancy and requires prompt management. Most of the cases have been reported to develop from epidermal cyst, chronic hydrocele or epididymis. This malignancy can lead to unexpected phenomena such as gastrointestinal bleeding or intestinal obstruction due to its unique metastatic pattern.