- Case Report
- Open Access
A metastasized hepatocellular carcinoma in the capsule of an undescended testis in the right inguinal area: report of a rare case
© The Author(s). 2018
- Received: 29 August 2017
- Accepted: 16 January 2018
- Published: 19 January 2018
Hepatocellular Carcinoma (HCC) is the most common primary carcinoma of the liver, which mainly metastasizes through the portal vein system.
Here, we report an extremely rare case in which HCC metastasized to the capsule of an undescended testis in the right inguinal area of the patient. A tumor approximately 8.8 × 7.0 cm in size was found in the patient’s liver during a health check-up. Initially, it was considered a metastatic tumor because the patient was found to have cryptorchidism, which had been left untreated before he presented to our hospital. The patient underwent a radical orchiectomy via inguinal approach, and the resected testis in the right inguinal region was examined via microscopy. The cancer cells were arranged in nests and showed abundant red or clear cytoplasm and marked nuclear atypia. Immunohistochemical staining showed that the tumor cells were positive for CK, CK8/18, AFP, hepatocyte, GCP3, but negative for PLAP, CD10, CD30, CD34, and vimentin.
According to these findings, the tumor in the inguinal region was considered a metastatic HCC arising from the liver, rather than a seminoma that had originated in the undescended testis. We suggest that during the diagnosis of malignancies, metastatic tumors should always be considered in the differential diagnosis even if the original presentation is at rare metastatic sites or concurrent with other disease(s).
- Hepatocellular carcinoma
- Undescended testis
- Inguinal region
- Case report
Hepatocellular carcinoma (HCC) is the most common primary carcinoma of the liver. The most common route by which hepatocellular cell carcinoma metastasizes is through the portal vein system. Distant hematogenous metastases are most frequently seen in the lungs , whereas rare distant metastatic sites include the jaw, skull, skin, and mandible [2–5]. Here, we report a rare case of HCC that had metastasized to the capsule of an undescended testis in the right inguinal region of a patient with cryptorchidism. The finding of HCC with histologically and immunohistochemically confirmed metastasis to a cryptorchid testis is itself a rarity. In the literature, there has been an isolated case of HCC with metastasis to the testis ; however, to the best of our knowledge, there has been no similar finding involving an undescended testis. Seminomas are more frequently found in the undescended testis , and can metastasize to the liver. As the patient had been found to have cryptorchidism before the mass in the liver was observed during a health check-up, the mass was initially considered to be a metastatic tumor from the undescended testis by the examining clinician. However, visualization via microscopy of the undescended testis showed a tumor present in the capsule, which is in accordance with the histological features of HCC. Thus, it is appropriate to consider the mass as a metastasized HCC from the liver, though it is an extremely rare presentation of the disease.
A 59-year-old man presented to our facility owing to an incidental finding of a liver mass during a routine health check-up a month earlier. He also had a cryptorchid right testis since birth. However, he was asymptomatic. Blood testing determined that α-fetoprotein (AFP) levels were markedly elevated (> 1210 ng/mL). The carcinoembryonic antigen (CEA) level was also slightly elevated (6.01 ng/mL). The levels of carbohydrate antigen 19–9 (CA19–9) and carbohydrate antigen 125 (CA125) were deemed to be within normal ranges. β-HCG was normal (2.3 mIU/ml).
The patient was treated with chemotherapy (5-FU + DDP). No more metastasis was detected 6 months after the therapy. AFP level reduced markedly after the chemotherapy (< 500 ng/mL). The tumor shrunk to 6.8 × 6.1 cm in size. The tumor metastasizing to the inguinal area was totally resected and had no relapse.
This study was approved by the institutional Ethics Committees of China Medical University and conducted in accordance with the ethical guidelines of the Declaration of Helsinki.
HCC is a common malignancy worldwide and the incidence has been rising (from 1.6/100000 to 4.6/100000) . Here, we report a case of metastatic HCC presenting in an extremely rare site in a patient who had a unique clinical history. The patient was found to have cryptorchidism and the undescended testis in his right inguinal region was not resected prior to reporting to our hospital. Cryptorchidism is found in roughly 1–3% of full-term newborns  and approximately 1% of 1-year-olds . The undescended testicles stay in the inguinal region or in the abdominal cavity. The risk of germ cell tumors increases in people with cryptorchidism compared to those without the disease . Seminoma is the most common type of germ cell tumor in the testes . The most common sites of hematogeneous spread of seminoma include the liver, lung, and bones . In this case, the tumor in the liver was originally considered a metastatic tumor from the undescended testis. Guo reported a case of seminoma which metastasized to the liver and neck . In this case, the patient underwent an orchidectomy of the testis in his right inguinal region. The tumor was located in the capsule of the testis and the histological and immunostaining findings proved the mass to be a metastatic HCC but not seminoma. The tumor cells were positive for AFP, a useful marker for early tumor detection of hepatocellular carcinoma. However, this marker is usually negative in seminoma . GPC-3 and hepatocyte are both specific markers for hepatocellular carcinoma, which were also positive in the tumor cells in the current case. On the contrary, PLAP is usually positive in seminoma but not hepatocellular carcinoma and was negative in the current case. These findings all support the diagnosis of hepatocellular carcinoma.
HCC mostly invades via the portal vein system and spreads in the liver . Distant metastases are mostly found in the lungs . Rare sites of distant metastases include the jaw, skull, skin, and mandible to name a few [2–5]. HCCs that metastasize to the testis are also rather rare. Young reported a case of HCC which metastasized to the testis in a 57-year-old man . Yamauchi reported a case of double cancer (hepatocellular carcinoma and mixed germ cell tumor of the testis) in a 43-year-old male . In this case, the HCC metastasized to the inguinal area adjacent to the undescended testis and it was located in the capsule of the testis but not inside the testis. Cancer cells may be involved in the inguinal area after invading the peritoneum first. HCC metastasis to the inguinal area is also rare and only one case of an HCC that had metastasized to the spermatic cord of the inguinal region has been reported . In this particular case, the HCC also metastasized to the inguinal region, where the undescended testis co-localized. There was not a clear mass in the testis, and only an abnormal signal in the capsule was detected via ultrasound. A few cancer cell nests were detected under the microscope in the capsule adjacent to the undescended testis. The histological findings of the testis were consistent with the features of an undescended testis. However, no seminoma was detected in the testis. A high serum AFP level is useful for predicting of HCC . In this case, the AFP level of the patient was markedly elevated (> 1210 ng/mL). Korenbaum reported an unusual case of hepatocellular carcinoma which was a second cancer after radiotherapy for testicular seminoma . In this case, the patient had not received any chemotherapy before the tumor in the liver was detected. According to the results of these findings, the tumor in the capsule of the undescended testis was determined to be a metastasized HCC, rather than seminoma, which can metastasize to the liver. Retroperitoneal lymph nodes, lung, and liver are the most common site of the metastasis of testicular seminoma [17–19]. There were also some complicated and unusual conditions related to testicular seminoma reported. Akin reported an extremely rare case of testicular seminoma accompanied by Wilson disease. Wilson disease caused liver cirrhosis which was suspected as metastasis of seminoma in the patient . Tumor-to-tumor metastases have also been documented. Ro reported a rare case of lung carcinoma which metastasized to testicular seminoma . Thus, the patients need a thorough examination to exclude the possibility of other tumors when metastasis of testicular seminoma is suspected. The cryptorchidism in this case is unilateral. Salman’s study indicated that the undescended testis impeded the germinal epithelium maturity in the contralateral descended testicle in mice . Another paper indicated that orchiectomy for unilateral cryptorchidism was helpful for the tissue development of the contralateral testis in mice . But, as no mass was detected by ultrasonography in the descended testis in the current case, pathological examination was not performed on it. However, the ultrasonography showed a relatively smaller testis in the left scrotum, which indicates possible hypoplasia of the testis.
HCCs that metastasize to the inguinal region or testis are both rather rare. Here, we report an exceptionally rare case in which an HCC metastasized to the capsule of the undescended testis located in the inguinal region. This exceedingly rare case illustrates the fact that during the diagnosis of malignancies, metastatic tumors should always be considered in the differential diagnosis even if they present in rare metastatic sites or concurrently with other disease(s).
This work was supported by the National Natural Science Foundation of China (no. 81472599 to Chuifeng Fan, MD).
Availability of data and materials
All data generated or analyzed during this study are included in this published article.
FCF and LQH performed the histopathological evaluation, literature review, and drafted the manuscript. FCF, LQH, and SXY performed the immunohistochemical stain evaluation. FCF conceived and designed the study. FCF gave the final histopathological diagnosis and revised the manuscript. All the authors read and approved the final manuscript.
Ethics approval and consent to participate
The institutional Ethics Committees of China Medical University approved the study.
Consent for publication
Written informed consent was obtained from the patient for publication of this case report and accompanying images.
The authors declare that they have no competing interests.
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- Bosman FT, Carneiro F, Hruban RH, Theise ND. WHO classification of tumors of the digestive system. 4th ed. Lyon: IRAC; 2015.Google Scholar
- Pesis M, Taicher S, Greenberg G, Hirshberg A. Metastasis to the jaws as a first manifestation of hepatocellular carcinoma: report of a case and analysis of 41 cases. J Craniomaxillofac Surg. 2014;42(8):1997–2001.View ArticlePubMedGoogle Scholar
- Cho E, Kim HS, Park YM, Kim HO, Lee JY. Cutaneous metastasis of hepatocellular carcinoma following skin injury after transcatheter arterial chemoembolization. Ann Dermatol. 2013;25(1):107–9.View ArticlePubMedPubMed CentralGoogle Scholar
- Chye CL, Lin KH, Ou CH, Sun CK, Chang IW, Liang CL. Acute spontaneous subdural hematoma caused by skull metastasis of hepatocellular carcinoma: case report. BMC Surg. 2015;15:60.View ArticlePubMedPubMed CentralGoogle Scholar
- Fujihara H, Chikazu D, Saijo H, Suenaga H, Mori Y, Iino M, Hamada Y, Takato T. Metastasis of hepatocellular carcinoma into the mandible with radiographic findings mimicking a radicular cyst: a case report. J Endod. 2010;36(9):1593–6.View ArticlePubMedGoogle Scholar
- Young RH, Van Patter HT, Scully RE. Hepatocellular carcinoma metastatic to the testis. Am J Clin Pathol. 1987;87(1):117–20.View ArticlePubMedGoogle Scholar
- Eble JN, Sauter G, Epstein JI, Sesterhenn IA. WHO classification of tumors of the digestive system. 3rd ed. Lyon: IRAC; 2004.Google Scholar
- Balogh J, Victor D 3rd, Asham EH, Burroughs SG, Boktour M, Saharia A, Li X, Ghobrial RM, Monsour HP Jr. Hepatocellular carcinoma: a review. J Hepatocell Carcinoma. 2016;3:41–53.View ArticlePubMedPubMed CentralGoogle Scholar
- Cobellis G, Noviello C, Nino F, Romano M, Mariscoli F, Martino A, Parmeggiani P, Papparella A. Spermatogenesis and cryptorchidism. Front Endocrinol (Lausanne). 2014;5:63.Google Scholar
- Barthold JS, González R. The epidemiology of congenital cryptorchidism, testicular ascent and orchiopexy. J Urol. 2003;170(6 Pt 1):2396–401.View ArticlePubMedGoogle Scholar
- Lee PA, Houk CP. Cryptorchidism. Curr Opin Endocrinol Diabetes Obes. 2013;20(3):210–6.View ArticlePubMedGoogle Scholar
- Guo KM, Liu Y, Zhong YP, Wang HL. Giant seminoma in an undescended testicle metastasizing to the neck and liver. Mol Clin Oncol. 2016;4(6):983–5.View ArticlePubMedPubMed CentralGoogle Scholar
- Eble JN, Sauter G, Epstein JI, Sesterhenn IA. WHO classification of tumors of the urinary system and male genital organs. 3rd ed. Lyon: IRAC; 2004.Google Scholar
- Yamauchi M, Yoshigoe F, Mera F, Ogura K, Kameda H, Nakada J, Takasaka S, Machida T, Fukunaga M. An autopsy case of double cancer (Hepatocellular carcinoma and mixed germ cell tumor of the testis)--significance of alpha-fetoprotein and human chorionic gonadotropin as tumor markers. Gan No Rinsho. 1983;29(4):A-22. 349-352Google Scholar
- Chiang HC, Chen PH, Shih HJ. Spermatic cord metastasis of primary hepatocellular carcinoma presenting as an inguinal mass: a case report. ISRN Oncol. 2011;2011:612753.PubMedPubMed CentralGoogle Scholar
- Korenbaum C, Barthelemy P, Onea A, Salze P, Kurtz JE. “Hockey stick” may strike back: Hepatocellular carcinoma on noncirrhotic liver as a late toxicity of lombo-aortic radiotherapy for seminoma. A review triggered by an unusual case. J Clin Exp Hepatol. 2016;6(1):54–8.View ArticlePubMedGoogle Scholar
- Johnson DE, Appelt G, Samuels ML, Luna M. Metastases from testicular carcinoma. Study of 78 autopsied cases. Urology. 1976 Sep;8(3):234–9.View ArticlePubMedGoogle Scholar
- Althaf S, Shankar K, Kurpad V, Suma MN. Seminoma of undescended testis with urinary bladder metastasis: a case report with review of literature. Urol Ann. 2017;9(1):77–9.View ArticlePubMedPubMed CentralGoogle Scholar
- Ferri E, Pedruzzi B, Gasparin P, Lunghi M. Cervical mass as the first clinical manifestation of unsuspected metastatic seminoma. Auris Nasus Larynx. 2017. doi:https://doi.org/10.1016/j.anl.2017.07.015. [Epub ahead of print].
- Akin M, Aksakal G, Songür Y, Işler M, Akin M. From suspicion of liver metastases to the diagnosis of Wilson disease in a patient with seminoma. Turk J Gastroenterol. 2012;23(2):165–8.View ArticlePubMedGoogle Scholar
- Ro JY, Sahin AA, Ayala AG, Ordonez NG, Grignon DJ, Popok SM. Lung carcinoma with metastasis to testicular seminoma. Cancer. 1990;66(2):347–53.View ArticlePubMedGoogle Scholar
- Salman FT, Adkins ES, Fonkalsrud EW. Morphologic effects of unilateral cryptorchidism on the contralateral descended testis. J Pediatr Surg. 1988;23(5):439–43.View ArticlePubMedGoogle Scholar
- Salman FT, Adkins ES, Fonkalsrud EW. Morphologic effects of orchiopexy or orchiectomy on the contralateral testis in experimental unilateral cryptorchidism. Surgery. 1988;103(5):573–8.PubMedGoogle Scholar