A Small Cell Carcinoma in the Testis Associated with Testicular Teratoma

Abstract

Dear Editor, Neuroendocrine neoplasms of the testis are extremely rare, accounting for 0.2% of all testicular neoplasms. Although the origin of the primary testicular neuroendocrine tumour is controversial, components of mature teratoma, leydig cells and intratubular germ cell neoplasia have been reported as precursor lesions (1, 2). Although a case of small cell carcinoma (SmCC) originating from an ovarian teratoma has been reported (3), to our knowledge there are no reports in English including SmCC of the testis originating from a testicular teratoma. In this paper, we present a metastatic SmCC of the testicle associated with testicular teratoma, along with its pathological properties, clinical course and treatment results.A 44-year-old male patient with a painless testicular mass underwent left radical orchiectomy in another hospital in 2010. The histopathological diagnosis was a 12 cm mixed germ cell tumour (GCT) composed of mature teratoma and embryonic cell carcinoma. Beta-human-chorionic-gonadotropin, alpha-fetoprotein levels and lactate-dehydrogenase levels were normal. Multiple pulmonary nodules (the largest 6 cm) and paraaortic lymphadenopathy (the largest 1.5 cm) were detected by computed tomography (CT).The patient was relatively old for non-teratoma non-seminoma GCT and tumour markers were normal; thus the orchiectomy material was reexamined in our centre to exclude other neoplasms besides GCT. Immunohistochemical and morphological findings were consistent with SmCC originating from mature teratoma (chromogranin, synaptophysin, neuron-specific enolase positive, CD56, MIC-2, GFAP negative).Although the patient had a diagnosis of SmCC, we treated him according to the principles of GCT treatment. Based on the knowledge that extrapulmonary SmCCs may behave in a different way than pulmonary SmCCs in terms of the organ of origin, we opted for germ cell carcinoma treatment rather than classical SmCC. As the chemotherapy protocol (cisplatin/ etoposide/bleomycin) that is effective against GCT involves similar agents and doses as the cisplatin/etoposide protocol used against SmCC, we applied four cycles of cisplatin/etoposide/bleomycin. After partial response had been achieved, two additional cycles of cisplatin/etoposide were applied (to protect against bleomycin toxicity). After six courses of chemotherapy, the paraaortic lympadenopathy in the abdominal CT disappeared and a large number of lesions disappeared on the thorax CT, but three lesions (the largest 2 cm) remained. Three months after the completion of chemotherapy, pathological FDG involvement was not observed in residual pulmonary lesions on PET/CT.In accordance with the GCT treatment approach, metastasectomy oriented to teratoma and microscopic residual disease was planned. However, the patient declined surgery. The patient has since been followed without recurrence and metastasis for 15 months. Although it was not possible to apply surgical treatment oriented to lung metastasis in our case, the efficiency of our treatment led us to believe that ...

Metastatic Primary Testicular Neuroendocrine Carcinoma Associated with Somatic Malignant Transformation of Teratoma: A Rare Case Report

“…The most frequently transformed histologic types are rhabdomyosarcoma, adenocarcinoma, and PNET, whereas the NET subtype is rare [ 5 ]. To date, only 24 cases of TNET arising in testicular teratoma have been reported, and only 2 of these cases involved visceral metastasis [ 8 , 9 , 10 ].…”

“…In another report on 2 TNET cases with lung metastasis, the tumor sizes were 12.0 and 10.8 cm. Of note, small-cell carcinoma transformation was reported in one of them [ 9 , 10 ]. Taken together, teratoma with SMT, especially with high-grade malignant histology, should always be suspected in the case of a young male presenting with rapid growth of a huge testicular mass with typical imaging findings for testicular teratoma, particularly with metastatic disease.…”

“…So far, there are only 2 other case reports in the literature concerning patients with TNET associated with teratoma involving visceral metastasis. Türkmen et al [ 9 ] reported a 44-year-old man with metastatic small-cell carcinoma associated with mature testicular teratoma and embryonal-cell carcinoma involving lung and lymph node metastasis. There was no information on Ki-67 reported.…”

“…Interestingly, no patient developed visceral metastases [ 8 ]. Until now, only 2 other cases of TNET arising from teratoma with visceral metastasis to the lungs have been reported in the English literature [ 9 , 10 ]. Of note, one of those cases showed small-cell carcinoma histology.…”

Extrapulmonary small cell carcinoma. Literature review

“…Встречаются смешанные опухоли. Представлен случай успешного лечения комбинацией цисплатин + этопозид + блеомицин [59]. Эта комбинация обсуждается для лечения МР яичка, так как она используется при герминогенных опухолях яичка.…”