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Testicular dysgenesis with the neoplastic lesion in an intersexual man: observation and clinical procedures from neonatal period to 29 year of life
Article published in Urologia Polska 2005/58/2.


Jolanta Słowikowska-Hilczer 1, Maria Szarras-Czapnik 2, Marek Sosnowski 3, Elżbieta Oszukowska 1, Jan Karol Wolski 4, Tomasz Romer 2, Krzysztof Kula 1
1 Katedra Andrologii i Endokrynologii Płodności, Uniwersytet Medyczny w Łodzi
Kierownik katedry: prof. dr hab. Krzysztof Kula
2 Klinika Pediatrii, Oddział Endokrynologii, Instytut ?Pomnik - Centrum Zdrowia Dziecka? w Warszawie
Kierownik kliniki: doc dr hab. Janusz Książyk
3 Klinika Urologii i Rehabilitacji Urologicznej, Uniwersytet Medyczny w Łodzi
Kierownik kliniki: prof. dr hab. Marek Sosnowski
4 Klinika Urologii CMKP w Warszawie
Kierownik kliniki: prof. dr hab. Andrzej Borówka


gonadal dysgenesis, intratubular testicular carcinoma, hypogonadism, radiotherapy, hormonal substitution


In a newborn child with ambiguous genitalia and gonads located in the minor abdomen cavity 45, X/46, XY karyotype was found. He was reared as a man. When the patient was 10-year-old one smaller gonad was removed. Histopathological examination revealed testicular structure with the presence of Sertoli cells and abnormal germ cells considered as gonocytes or CIS cells, but CIS was not diagnosed because of the lack of the specific antigen placental-like alkaline phosphatase (PLAP). Puberty started and progressed in time with normal hormone levels in the blood (testosterone 12.6 nmol/l; LH 3.4 IU/l). However, selectively increased blood level of FSH (26.0 IU/l) indicated the lesion of the seminal tubules function. At the age of 25 years the biopsy of the preserved testis was performed. Abnormal germ cells were positive for PLAP and CIS was diagnosed. The patient received a course of irradiation on the testis. In the biopsy performed 3 years after the treatment CIS was not found, tubules contained only Sertoli cells. Blood level of testosterone decreased to 5.3 nmol/l together with the increase of gonadotropins (FSH 54.4 IU/l; LH 36.5 IU/l). Hormonal substitution with testosterone was introduced and, when terminated after 2 years, testosterone secretion recovered to the pre-radiotherapy values.
These observations show that despite preserved endocrine function dysgenetic testes of the adult man may be burden with the preinvasive germ cell cancer since childhood. Histopathological examination towards CIS guarantees early and effective treatment. After radiotherapy because of CIS testicular endocrine function may decrease, what needs hormonal substitution, but after longer time endocrine function may improve spontaneously.


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Jolanta Słowikowska-Hilczer
Katedra Andrologii i Endokrynologii Płodności
Uniwersytet Medyczny w Łodzi
ul. Sterlinga 5
91-425 Łódź
tel./fax (042) 633 07 05