Antonio Leen, Albersen Maarten, Billen Jaak, Maleux Geert, Van Rompuy Anne-Sophie, Coremans Peter, Marcq Philippe, Jørgensen Niels, Vanderschueren Dirk
Department of Endocrinology, University Hospitals Leuven, Leuven, Belgium.
Department of Chronic Diseases, Metabolism and Ageing, Laboratory of Clinical and Experimental Endocrinology, KU Leuven, Leuven, Belgium.
J Endocr Soc. 2019 Jul 30;3(10):1881-1886. doi: 10.1210/js.2019-00180. eCollection 2019 Oct 1.
Suppressed gonadotropins combined with high-normal serum testosterone concentrations in oligozoospermic men suggest either use of exogenous testosterone or presence of a testosterone-producing tumor. We describe the case of a 31-year-old man referred for primary infertility. Gonadotropins were undetectably low, but testosterone and estradiol were in the high-normal range. Semen analysis showed oligoasthenospermia. He denied using exogenous testosterone. Scrotal ultrasound showed microlithiasis and millimetric hypolucent lesions in the left testis but no intratesticular mass. Human chorionic gonadotropin was low. To investigate unilateral hormone secretion, selective testicular venous sampling was performed. Testosterone and estradiol were clearly higher on the left side than on the right (130 vs 26 nmol/L and 1388 vs 62 pmol/L, respectively), with a left spermatic vein-to-periphery gradient of 4.3 for testosterone and 13 for estradiol; there were no similar gradients on the right side. This finding confirms that all sex steroid secretion came from the left testis. The patient was therefore referred for left orchidectomy. Histopathology revealed multifocal seminoma, germ cell neoplasia and Leydig cell hyperplasia but no choriocarcinoma. However, gonadotrophin levels increased after orchidectomy, indicating that the source of gonadotropin-independent sex steroid secretion was removed. Testosterone and estradiol decreased to the mid-normal range. Sperm concentration improved. This report thus shows that endogenous testosterone secretion in one testicle supports spermatogenesis without measurable levels of gonadotropins. Selective testicular venous sampling is useful to identify the site of unilateral secretion when the clinical picture is inconclusive. However, histopathology could not reveal the factor that stimulated Leydig cell steroidogenesis.
少精子症男性中促性腺激素水平受抑制,同时血清睾酮浓度处于正常高值,提示可能使用了外源性睾酮或存在分泌睾酮的肿瘤。我们描述了一名因原发性不育前来就诊的31岁男性病例。促性腺激素水平低至检测不到,但睾酮和雌二醇处于正常高值范围。精液分析显示少弱精子症。他否认使用过外源性睾酮。阴囊超声显示左侧睾丸有微结石和毫米级低回声病变,但无睾丸内肿块。人绒毛膜促性腺激素水平较低。为了研究单侧激素分泌情况,进行了选择性睾丸静脉采血。左侧睾酮和雌二醇水平明显高于右侧(分别为130 vs 26 nmol/L和1388 vs 62 pmol/L),左侧精索静脉与外周的睾酮梯度为4.3,雌二醇梯度为13;右侧无类似梯度。这一发现证实所有性类固醇分泌均来自左侧睾丸。因此,该患者被转诊进行左侧睾丸切除术。组织病理学检查显示多灶性精原细胞瘤、生殖细胞肿瘤和Leydig细胞增生,但无绒毛膜癌。然而,睾丸切除术后促性腺激素水平升高,表明非促性腺激素依赖性性类固醇分泌的来源已被切除。睾酮和雌二醇降至正常范围的中间值。精子浓度有所改善。本报告表明,一侧睾丸内源性睾酮分泌在促性腺激素水平无法检测时仍支持精子发生。当临床表现不明确时,选择性睾丸静脉采血有助于确定单侧分泌的部位。然而,组织病理学未能揭示刺激Leydig细胞类固醇生成的因素。