Leydig cell tumor of the testis with azoospermia and elevated delta4 androstenedione: Case report

被引:4
作者
Prasivoravong J. [1 ,6 ]
Barbotin A.-L. [2 ,5 ]
Derveaux A. [1 ]
Leroy C. [1 ]
Leroy X. [3 ]
Puech P. [4 ]
Mitchell V. [2 ,5 ]
Marcelli F. [1 ,5 ]
Rigot J.-M. [1 ,5 ]
机构
[1] Lille University Hospital, Department of Andrology, Lille
[2] Lille University Hospital, Biology of Reproduction Unit, Lille
[3] Lille University Hospital, Department of Pathology, Lille
[4] Lille University Hospital, Department of Radiology, Lille
[5] University of Lille, EA4308 Gametogenesis and Gamete Quality, Lille
[6] Hôpital Calmette, Department of Andrology, CHRU Lille, Boulevard du Professeur Leclercq, Lille Cedex
关键词
Azoospermia; Delta4; androstenedione; Hormone secreting testicular tumor; Infertility; Leydig cell tumor; TESE;
D O I
10.1186/s12610-016-0041-8
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学科分类号
摘要
Background: Secreting interstitial cell (Leydig cell) tumors are rare. In adults, the clinical picture and steroid levels are variable. Case presentation: 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. Conclusions: In this case, the diagnosis of testicular Leydig cell tumor secreting delta4 androstenedione was made in a context of azoospermia. © 2016 The Author(s).
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  • [1] Turner W.R., Derrick F.C., Wohltmann H., Leydig cell tumor in identical twin, Urology, 7, 2, pp. 194-197, (1976)
  • [2] Boulanger P., Somma M., Chevalier S., Bleau G., Roberts K.D., Chapdelaine A., Elevated secretion of androstenedione in a patient with a Leydig cell tumour, Acta Endocrinol (Copenh), 107, 1, pp. 104-109, (1984)
  • [3] Kondoh N., Koh E., Nakamura M., Namiki M., Kiyohara H., Okuyama A., Et al., Bilateral Leydig cell tumors and male infertility: case report, Urol Int, 46, 1, pp. 104-106, (1991)
  • [4] Gabrilove J.L., Nicolis G.L., Mitty H.A., Sohval A.R., Feminizing interstitial cell tumor of the testis: personal observations and a review of the literature, Cancer, 35, 4, pp. 1184-1202, (1975)
  • [5] Perez C., Novoa J., Alcaniz J., Salto L., Barcelo B., Leydig cell tumour of the testis with gynaecomastia and elevated oestrogen, progesterone and prolactin levels: case report, Clin Endocrinol (Oxf), 13, 5, pp. 409-412, (1980)
  • [6] Mineur P., Cooman S., Hustin J., Verhoeven G., Hertogh R., Feminizing testicular Leydig cell tumor: hormonal profile before and after unilateral orchidectomy, J Clin Endocrinol Metab, 64, 4, pp. 686-691, (1987)
  • [7] Valensi P., Coussieu C., Kemeny J.L., Attali J.R., Amouroux J., Sebaoun J., Endocrine investigations in two cases of feminizing Leydig cell tumour, Acta Endocrinol (Copenh), 115, 3, pp. 365-372, (1987)
  • [8] Schwarzman M.I., Russo P., Bosl G.J., Whitmore W.F., Hormone-secreting metastatic interstitial cell tumor of the testis, J Urol, 141, 3, pp. 620-622, (1989)
  • [9] Kerlan V., Nahoul K., Abalain J.H., Mangin P., Bercovici J.P., Oestrogen secreting Leydig cell tumour and GnRH agonist in-vivo and in-vitro studies, Clin Endocrinol (Oxf), 37, 3, pp. 221-226, (1992)
  • [10] Caron P.J., Bennet A.P., Plantavid M.M., Louvet J.P., Luteinizing hormone secretory pattern before and after removal of Leydig cell tumor of the testis, Eur J Endocrinol Eur Fed Endocr Soc, 131, 2, pp. 156-159, (1994)