A 37-year-old man presented with exhaustion and reduced strength in his right arm. Physical examination showed marked anemia and a hard, large tumor of the left testicle, which the patient had been observing for several months. There was a history of extensive cocaine and nicotine consumption. Scrotal ultrasound confirmed a large testicular tumor. Serum tumor markers were increased (Α-fetoprotein [AFP] 15.9 ng/ml [<5.8 ng/ml], Β-human chorion gonadotropin [Β-HCG] 128,292 IU/ml [<2 IU/ml] and lactate dehydrogenase 363 U/L [<250 U/L]). Computerized tomography scan of the thorax, abdomen and pelvis showed multiple metastases in the retroperitoneal lymph nodes, liver and lungs. Magnetic resonance imaging (MRI) of the head showed 3 supra- and infratentorial lesions with pronounced perifocal edema and displacement of the hemispherical gap.

The patient was offered sperm cryoconservation and systemic chemotherapy with cisplatin, etoposide and ifosfamide (PEI) was started, with a dose reduction during the first cycle. With chemotherapy, there was a marked improvement of the neurological symptoms. Blood transfusions were given, but anemia persisted. Further evaluation showed iron deficiency anemia, and the patient also reported repeated rectal blood discharge. Upper gastrointestinal endoscopy was normal, but on colonoscopy an ulcerated tumor of the ileocecal valve was seen (fig. 1). Biopsy confirmed a poorly differentiated, pleomorphic cell carcinoma with mononuclear and multinucleated giant cells and very strong expression of ß-HCG (fig. 2).

Figure 1. Cause of anemia requiring transfusion with recurrent rectal blood outflows: metastasis of a nonseminoma in the area of the ileocecal valve.

A total of 4 cycles of PEI polychemotherapy were given and well tolerated. Under chemotherapy, the rectal bleeding stopped, the hemoglobin level normalized and the patient had better appetite and gained 17 kg in body weight. The reduction in serum tumor markers was adequate (AFP 1.7 ng/ml, Β-HCG 110 IU/ml, lactate dehydrogenase 264 U/L). After the 4 cycles, computerized tomography scanning showed a clear regression of the intra-abdominal and thoracic metastases. Repeat colonoscopy could no longer detect the previously seen tumor of the ileocecal valve.

Figure 2. Peritoneal metastasis of a nonseminomatous germ cell tumor with trophoblastic and syncytiotrophoblastic differentiation. A, hematoxillin-eosin stained peritoneal tissue biopsy showed a biphasic dysplastic tumor cell population consisting of mononucleated trophoblastic and multinucleated syncytiotrophoblastic cells in an inflammatory and necrotic background (H&E, 10×). B, tumor cells displayed no relevant Pas reactivity (Peroxidase-Schiff-reagent, 10×). C, mononuclear and multinucleated dysplastic cells expressed cytokeratin (AE1/3, 20×), and D, were strongly ß-HCG immunoreactive, but completely immunonegative for placental alkaline phosphatase, CD30, AFP and OCT3/4 (ß-human chorionic gonadotropin, 10×).

Then the left testicle was surgically removed and a still large, but largely devitalized, germ cell tumor was seen with the only still vital tumor tissue described as mature teratoma. Cerebral MRI did, however, show only a mixed treatment response. There was stable disease noted in the previously seen cerebral lesions, but both supra- and infratentorially, several new, small metastases were detected (fig. 3). After extensive multidisciplinary discussions, high-dose chemotherapy of PEI with subsequent autologous stem cell transplantation and additive stereotactical radiotherapy of the cerebral metastases were started. For this purpose, blood stem cells were taken from the patient before chemotherapy, which will be transferred after the treatment is completed. High-dose chemotherapy is currently only performed in special centers.

Discussion

Figure 3. Cerebral MRI noted in the previously seen cerebral lesions, but both supra- and infratentorially, several new, small metastases were detected.

This case is unique in that intermittent rectal bleeding occurred in metastatic testicular tumor disease caused by an ileocecal valve metastasis leading to marked iron-deficiency anemia. Intestinal metastasis of testicular cancer is rare and is seen in less than 5% of germ cell tumors.^1,2 It occurs more often with nonseminomas and especially with embryonal carcinoma. Presumably this ileocecal valve metastasis arose from direct spread from the retroperitoneal lymph node metastases.^1,3–6 Gastrointestinal metastases of germ-cell tumors do not occur without other widespread metastatic disease and can lead to bleeding with anemia as in this patient, but also to gastric pain, intestinal obstruction, perforation or even intussusception.^2,6 First line treatment for gastrointestinal tract metastases is systemic chemotherapy.^7,8 Furthermore, this case again illustrates the devastating effect of treatment delay, as this patient had observed his increasing testicular tumor for many months.

Clinical Practice Points

In patients with advanced testicular cancer, gastrointestinal symptoms can rarely be caused by gastrointestinal metastasis.