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      Cholangiocarcinoma presenting as a solitary epididymal metastasis: a case report and review of the literature

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      1 , , 2
      Diagnostic Pathology
      BioMed Central

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          Abstract

          Background

          Solid tumor metastasis to the epididymis is a rare occurrence and is mostly discovered incidentally at autopsy or after therapeutic orchidectomy for prostate cancer. Other primary carcinomas that have been demonstrated to metastasize to the paratesticular region include those originating in the stomach, kidney, ileum, and colon.

          Case presentation

          A 72-year-old gentleman presented with a firm and tender mass involving the right epididymis. On examination, he was jaundiced. Computed tomography of the abdomen demonstrated an obstructive stricture of the extra-hepatic bile ducts, in keeping with a cholangiocarcinoma, through which a metal stent was endoscopically inserted for symptomatic relief.

          Subsequent right radical orchidectomy yielded a diffusely infiltrative adenocarcinoma obliterating the epididymis, extending into the rete testis, vas deferens and spermatic cord and showing widespread vascular and perineural invasion. Residual epididymal, rete, and testicular tubules showed no in situ neoplasia. Morphologically and immunohistochemically the features were in keeping with a metastasis from a primary cholangiocarcinoma.

          Conclusion

          Only two cases of bile duct carcinoma metastasising to the male genital tract have previously been reported in the literature, the testis being the main site of metastasis in both cases. To our knowledge, this is the first described case of cholangiocarcinoma metastasising primarily to the epididymis, and presenting as a solitary epididymal metastasis in the absence of disseminated disease. It serves to highlight the importance of performing a thorough examination of the male external genitalia both clinically, in the follow up of cancer patients, and at autopsy.

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          Most cited references12

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          Selected other problematic testicular and paratesticular lesions: rete testis neoplasms and pseudotumors, mesothelial lesions and secondary tumors.

          The proximity and, in some instances, communication between several structures in the testis and paratestis (rete testis, epididymis, mesothelium, vestigial epithelium and paratesticular soft tissue) result in a plethora of interesting tumors and tumor-like lesions that together pose a formidable diagnostic challenge both because of their morphologic overlap and rarity. The occasional spread of tumors primarily at other sites to this region adds to the potential problem encountered. This review provides an overview of the pathology of nonmesenchymal paratesticular neoplasms and pseudotumors with a focus on the approach to tubulopapillary neoplasms for which diagnostic considerations may include carcinoma of the rete testis, malignant mesothelioma, ovarian-type epithelial tumors, epididymal carcinoma and metastatic carcinomas. The cornerstone of accurate characterization of these lesions is still a comprehensive, traditional clinicopathologic approach, clinical history (of another primary), gross examination (location) and routine light microscopy, but judicious incorporation of contemporary immunohistochemical markers may aid or in some instances be crucial in resolving the problems encountered.
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            Secondary neoplasms of the male genital tract with different patterns of involvement in adults and children.

            The incidence, presentation and macroscopic and histological features of secondary neoplasms of the male genital tract are described with reference to their differential diagnosis. A retrospective study of cases from the Royal London Hospital yielded a total of 31 secondary neoplasms involving the testis: 14 at postmortem examination and 17 surgical specimens. Nine cases were leukaemias: six acute lymphoblastic and two acute myeloid leukaemias in children, and one chronic lymphocytic leukaemia in an adult. The commonest primary sites of metastases to the testis were prostate (six cases), stomach (five cases) and lung (three cases). There were two malignant melanomas and isolated examples of metastases from the adrenal gland (neuroblastoma), cerebellum (medulloblastoma), soft tissue (alveolar rhabdomyosarcoma), pancreas and rectum. Of the metastases from solid tumours, 12 involved the right testis only, three involved the left and four were bilateral. In seven of these cases there were multiple testicular nodules, in seven there was a single mass, and in the rest there was diffuse involvement. Secondary neoplasms represented 4.6% of all testicular neoplasms at autopsy, and 1.6% in surgical specimens. There were five secondary penile neoplasms: two each from the pancreas and prostate and one from the bladder. Two neoplasms metastatic to the spermatic cord, both from a gastric primary, were included in the series. Secondary neoplasms of the testis occur with a frequency comparable to other sites in the genitourinary tract, and metastases to the spermatic cord, epididymis, and penis, are rare in comparison. Disseminated neoplasms rarely present initially at this site and are histologically distinctive in adults, but in children they must be distinguished from primary small round blue cell tumours.
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              Metastatic carcinoma involving the testis. Clinical and pathologic distinction from primary testicular neoplasms.

              Metastatic carcinoma to the testis is unusual. There are only seven previously reported cases in which a testicular mass was the first clinical manifestation of an underlying malignancy. The authors review 127 cases in which the testis was involved by metastatic carcinoma, and describe an additional two patients in whom a malignant testicular mass was the presenting sign of an underlying nontesticular carcinoma. The tumors most commonly reported to metastasize to the testis are: prostate (45 cases), lung (25 cases), melanoma (12 cases), colon (11 cases), kidney (10 cases), stomach (6 cases), and pancreas (5 cases). Neuroblastoma, retinoblastoma, carcinoid tumor, and cancers of the bile duct, ureter, bladder, salivary gland, and thyroid have also involved the testis secondarily. Nineteen patients (15%) had bilateral testicular metastases. Patients with secondary testicular neoplasms were older in general than those with germ cell tumors (mean, 55 years; median, 57 years). Histologically, the presence of extensive lymphatic and vascular invasion and an interstitial pattern, in which the seminiferous tubules are spared, is suggestive of a metastasis. In four of the nine cases (44%) in which testicular enlargement was the first manifestation of an underlying carcinoma the correct pathologic diagnosis was initially missed. Serum alpha-fetoprotein (AFP) and human chorionic gonadotropin (HCG) are occasionally elevated in patients with nontesticular primary tumors, but markedly elevated levels in young patients suggest a nonseminomatous germ cell tumor, as does positive immunoperoxidase staining for AFP and HCG.
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                Author and article information

                Journal
                Diagn Pathol
                Diagnostic Pathology
                BioMed Central (London )
                1746-1596
                2007
                30 August 2007
                : 2
                : 33
                Affiliations
                [1 ]Department of Cellular Pathology, John Radcliffe Hospital, Headley Way, Headington, Oxford, OX3 9DU, UK
                [2 ]Department of Cellular Pathology, Wycombe General Hospital, Queen Alexandra Road, High Wycombe, Buckinghamshire, HP11 2TT, UK
                Article
                1746-1596-2-33
                10.1186/1746-1596-2-33
                2000863
                17760973
                3339ccc8-b9ed-4b75-98aa-12288bdfadbc
                Copyright © 2007 Bennett and Bailey; licensee BioMed Central Ltd.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                : 24 July 2007
                : 30 August 2007
                Categories
                Case Report

                Pathology
                Pathology

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