Prasivoravong J, Barbotin A-L, Derveaux A, Leroy C, Leroy X, Puech P, Mitchell V, Marcelli F, Rigot J-M
Department of Andrology, Lille University Hospital, Lille, France ; Department of Andrology, CHRU Lille, Hôpital Calmette, Boulevard du Professeur Leclercq, 59037 Lille Cedex, France.
Biology of Reproduction Unit, Lille University Hospital, Lille, France ; EA4308 Gametogenesis and Gamete Quality, University of Lille, Lille, France.
Basic Clin Androl. 2016 Nov 8;26:14. doi: 10.1186/s12610-016-0041-8. eCollection 2016.
Secreting interstitial cell (Leydig cell) tumors are rare. In adults, the clinical picture and steroid levels are variable.
This paper presents a case of left testicular tumor, showing azoospermia with normal serum level of total testosterone, collapsed FSH and LH, and high delta4 androstenedione. Histopathological investigation revealed a Leydig cell tumor. TESE allowed spermatozoa extraction and freezing. Testicular histology found hypospermatogenesis and germ-cell aplasia with interstitial fibrosis. Surgical resection of the tumor resulted in normalization of gonadotropins and fall in serum delta4 androstenedione to subnormal levels in the postoperative period confirming that the tumor was secreting delta4 androstenedione. It was hypothesized that high delta4 androstenedione resulted in intra tumoral 17 β-HSD overtaken by delta4 androstenedione or that 17 β-HSD activity in the tumor was different from that of normal Leydig cells. Three months after surgery sperm analysis found a complete recovery of spermatogenesis. A spontaneous pregnancy occurred 3 months after surgery and a girl was born.
In this case, the diagnosis of testicular Leydig cell tumor secreting delta4 androstenedione was made in a context of azoospermia.
分泌性间质细胞瘤（Leydig细胞瘤）较为罕见。在成年人中，临床表现和类固醇水平各不相同。
本文介绍了一例左侧睾丸肿瘤病例，患者表现为无精子症，血清总睾酮水平正常，促卵泡生成素（FSH）和促黄体生成素（LH）降低，以及δ4雄烯二酮升高。组织病理学检查显示为Leydig细胞瘤。睾丸精子提取术（TESE）成功提取并冷冻了精子。睾丸组织学检查发现精子发生减少和生精细胞发育不全伴间质纤维化。肿瘤手术切除后，促性腺激素恢复正常，术后血清δ4雄烯二酮降至正常水平以下，证实肿瘤分泌δ4雄烯二酮。推测δ4雄烯二酮升高是由于肿瘤内17β-羟类固醇脱氢酶（17β-HSD）被δ4雄烯二酮取代，或者肿瘤中的17β-HSD活性与正常Leydig细胞不同。术后三个月的精子分析发现精子发生完全恢复。术后三个月自然受孕并产下一名女婴。
在本病例中，在无精子症的背景下诊断出分泌δ4雄烯二酮的睾丸Leydig细胞瘤。
