Testicular cancers comprise 1-1.5% of entire cancers in men, and sex cord-stromal tumors include 5% of testicular cancers. This study aims to report a simultaneous Sertoli cell-only syndrome and Leydig cell tumor in the same patient. A 32-year-old man presented with a history of primary infertility for 3 years. Physical examination revealed normal secondary sexual characteristics. Two successive seminal fluid analyses revealed azoospermia. A scrotal ultrasound scan showed a 28 × 27 mm hypoechoic and hypervascular right testicular mass. Right radical orchiectomy and simultaneous left testicular biopsy were conducted. The histopathological examination revealed Sertoli cell-only syndrome and Leydig cell tumor with focal Leydig cell hyperplasia. Reversing fertility following the management of Leydig cell tumor is rarely mentioned in the literature. A study revealed that fertility recovered following 4 months of management in a primary infertile male. However, infertile men with nonobstructive azoospermia due to SCOS can only have a child by testicular sperm extraction technique. Despite the rare occurrence of Leydig cell tumor, it could be seen in association with Sertoli cell-only syndrome in infertile men with azoospermia. Clinical examination and imaging studies are important in these patients as the possibility of having a testicular mass is high among them.

We used fine needle aspiration cytology (FNAC) to diagnose Sertoli cell-only pattern and hypospermatogenesis in an Iberian red deer (Cervus elaphus hispanicus). Cytologic diagnosis was confirmed by histology and epididymal sperm analysis. We conclude that FNAC can be an important diagnostic tool in testicular diseases of wildlife.

Total Sertoli-cell-only (SCO) syndrome is often confused with a focal SCO picture, in which testicular illness caused damage to seminiferous tubules and compromised the Sertoli cell range of maturation and functions, but from which still some spermatozoa can be retrieved for assisted reproductive techniques. Here, a possibly new SCO syndrome phenotype is reported exhibiting complete lack of germ cells despite normal architecture of the seminiferous tubules with presence of mature Sertoli cells and normal Leydig cells in the intertubular tissue. Sertoli cells are immunonegative for the prepubertal differentiation markers cytokeratin-18, anti-Muellerian hormone and M2A antigen, but reveal a positive signal for the gap junctional protein connexin 43 known to be expressed in Sertoli cells with an adult type of differentiation. The complete lack of germ cells in combination with fully differentiated adult-type Sertoli cells in this case is in contradiction with known SCO subtypes and with the current hypothesis of reciprocal regulation of Sertoli and germ cell differentiation.

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