First reported case of paratesticular seminoma in a postpubertal cryptorchid testis

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Abstract

Cryptorchidism is a very common anomaly, affecting 2–4% of male infants and is more common in premature infants. The long-term outcome despite orchidopexy still remains problematic and controversial with a risk of developing cancer 5–10 times greater than normal. Paratesticular tumors are mostly benign and very rare in children however malignant paratesticular tumors do arise, the most common being rhabdomyosarcoma. Primary paratesticular seminoma is extremely rare by itself and in most cases is associated with foci of seminoma within the testis. To the best of our knowledge, our case represents the fourth reported case of paratesticular seminoma in the published literature and being the first one in cryptorchid testis.

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Cryptorchidism.

J Hutson, Tamara Nation, Adam Balic … (2010)

Cryptorchidism is a very common anomaly of the male genitalia, affecting 2%-4% of male infants and is more common in premature infants. There are two separate stages of testicular descent. The first stage occurs at 8-15 weeks' gestation in the human fetus and is characterized by enlargement of the genito-inguinal ligament, or gubernaculum, and regression of the cranial suspensory ligament. The testis remains close to the future inguinal region as the fetal abdomen grows. Leydig cells in the testis produce insulin-like hormone 3, which stimulates the caudal gubernaculum to grow and become thicker. Mullerian inhibiting substance may have a role in the first phase of descent by stimulating the swelling reaction in the gubernaculum. The second phase of testicular descent requires migration of the gubernaculum and testis from the inguinal region to the scrotum, between 25 and 35 weeks' gestation. The genitofemoral nerve releases calcitonin gene-related peptide, a neurotransmitter that provides a chemotactic gradient to guide migration. The exact cause of cyrptorchidism remains elusive. Information is mainly derived from animal studies (especially in rodents), which may not extrapolate to the human setting. These findings, however, do have some similarities among mammalian species. The current recommended timing for orchidopexy is between 6 and 12 months of life in an effort to preserve the spermatogonia--the stem cells for subsequent spermatogenesis. Despite surgical treatment by orchidopexy, the long-term outcome still remains problematic and controversial. Impaired fertility (33% in unilateral cases and 66% in bilateral undescended testes) and a cancer risk 5-10 times greater than normal is observed over time. Further research into the cause and management of undescended testes is necessary. Copyright 2010 Elsevier Inc. All rights reserved.

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What is new in cryptorchidism and hypospadias--a critical review on the testicular dysgenesis hypothesis.

Jorgen Thorup, Robert McLachlan, Dina Cortes … (2010)

It has been hypothesized that poor semen quality, testis cancer, undescended testis, and hypospadias are symptoms of one underlying entity--the testicular dysgenesis syndrome--leading to increasing male fertility impairment. Though testicular cancer has increased in many Western countries during the past 40 years, hypospadias rates have not changed with certainty over the same period. Also, recent studies demonstrate that sperm output may have declined in certain areas of Europe but is probably not declining across the globe as indicated by American studies. However, at the same time, there is increasing recognition of male infertility related to obesity and smoking. There is no certain evidence that the rates of undescended testes have been increasing with time during the last 50 years. In more than 95% of the cases, hypospadias is not associated with cryptorchidism, suggesting major differences in pathogenesis. Placental abnormality may occasionally cause both cryptorchidism and hypospadias, as it is also the case in many other congenital malformations. The findings of early orchidopexy lowering the risk of both infertility and testicular cancer suggest that the abnormal location exposes the cryptorchid testis to infertility and malignant transformation, rather than there being a primary abnormality. Statistically, 5% of testicular cancers only are caused by cryptorchidism. These data point to the complexity of pathogenic and epidemiologic features of each component and the difficulties in ascribing them to a single unifying process, such as testicular dysgenesis syndrome, particularly when so little is known of the actual mechanisms of disease. Copyright © 2010 Elsevier Inc. All rights reserved.

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Adult primary paratesticular mesenchymal tumors with emphasis on a case presentation and discussion of spermatic cord leiomyosarcoma

Andrea B Galosi, Marina Scarpelli, Roberta Mazzucchelli … (2014)

Background The aim of this report is related to adult primary paratesticular mesenchymal tumors with emphasis on a case presentation and discussion of the spermatic cord leiomyosarcoma. Primary paratesticular tumors are rare, only accounting for 7% to 10% of all intrascrotal tumors. In adults, more than 75% of these lesions arise from the spermatic cord, 20% being leiomyosarcoma. Tumor grade, stage, histologic type, and lymph node involvement are independently predictive of prognosis. Findings The case report concerns a 81-year-old man presented with a 3-year history of painless lump in the right hemiscrotum. Scrotal examination demonstrated a 5.1-cm, firm-to-hard mass attached to the spermatic cord. Scrotal ultrasound scan revealed a heterogeneous mass separate from the testis. He was treated with an radical orchi-funicolectomy. Histologically the lesion is composed of spindled cells with often elongated, blunt-ended nuclei and variably eosinophilic cytoplasm. Areas with pleomorphic morphology are present. The level of mitotic activity is equal to 3/10 HPF in the areas with spindle cell morphology and to 12/10 HPF in the areas with pleomorphic morphology. The final diagnosis was that a leiomyosarcoma of the spermatic cord, with grade 1 and grade 2 areas, stage pT2b cN0 and cM0. The patient has been followed up for 3 months with CT scans and shows no signs of recurrence. Conclusions Spermatic cord leiomyosarcoma, although rare, should be one of the first differential diagnoses for a firm-to-hard lump in the cord. Apart from radical orchi-funicolectomy, there has been added benefit of adjuvant radiotherapy to prevent any loco-regional lymph node recurrence. Virtual Slides The virtual slide(s) for this article can be found here: http://www.diagnosticpathology.diagnomx.eu/vs/1613030331125632

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Journal

Journal ID (nlm-ta): J Surg Case Rep

Journal ID (iso-abbrev): J Surg Case Rep

Journal ID (publisher-id): jscr

Title: Journal of Surgical Case Reports

Publisher: Oxford University Press

ISSN (Electronic): 2042-8812

Publication date Collection: June 2017

Publication date (Electronic): 20 June 2017

Publication date PMC-release: 20 June 2017

Volume: 2017

Issue: 6

Electronic Location Identifier: rjx088

Affiliations

[1 ]Department of Urology, Makassed General Hospital, Beirut, Lebanon

[2 ]Department of Oncology, Makassed General Hospital, Beirut, Lebanon

[3 ]Department of Surgery, Makassed General Hospital, Beirut, Lebanon

Author notes

[* ]Correspondence address. Makassed General Hospital, PO Box 11-6301 Riad EI-Solh, 11072210 Beirut, Lebanon. Tel: +961-163-6000; Personnel Phone number: +961-70-858-658; E-mail: dr.houssamabtar@ 123456gmail.com

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Publisher ID: rjx088

DOI: 10.1093/jscr/rjx088

PMC ID: 5499890

SO-VID: 6474bee8-90bb-46dd-bfa1-5bc433c92642

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This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License ( http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com

History

Date received : 26 February 2017

Date accepted : 09 June 2017

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Pages: 3

First reported case of paratesticular seminoma in a postpubertal cryptorchid testis

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Most cited references 10

Cryptorchidism.

What is new in cryptorchidism and hypospadias--a critical review on the testicular dysgenesis hypothesis.

Adult primary paratesticular mesenchymal tumors with emphasis on a case presentation and discussion of spermatic cord leiomyosarcoma

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