Occult leydig cell tumour and androgen-receptor positive breast cancer in a woman with severe hyperandrogenism

Journal of Ovarian Research

Salvatore Cannavo²  Francesco Trimarchi²  Valeria Barresi¹  Giovanna Saraceno² 

[1] Department of Pathology, University of Messina, Messina, Italy;Department of Clinical and Experimental Medicine, Via Consolare Valeria, Messina, 98125, Italy

关键词: Breast;    Androgen-receptor;    Ovary;    Polycythemia;    Hyperandrogenism;   

Others  :  810402
DOI  :  10.1186/1757-2215-6-43

 received in 2013-04-22, accepted in 2013-06-19,  发布年份 2013

【 摘 要 】

Leydig cell tumours represent more than 75% of all testosterone-secreting ovarian masses. These benign tumours are frequently occult or very small, but cause dramatic virilization. Chronic hyperandrogenism can also induce systemic complications, which increase morbidity and mortality risk. One of the most obvious effects of increased testosterone levels is polycythemia, a complication which induces dermatologic, osteoarticular and gastrointestinal manifestations and is associated with increased thrombotic risk. However, scientific literature reports few data concerning etiopathogenesis and management of polycythemia in patients with Leydig cell tumours. Moreover, no data are available about the effect of androgen excess on other concomitant tumours expressing androgen receptors. In this paper we report for the first time the case of a woman, with previous infertility, dramatic virilisation and chronic erythrocytosis, who was affected by an occult Leydig cell tumour and an androgen receptor positive breast cancer. This association gives us the opportunity to discuss the role of the steroid receptor expression of breast cancer in the presence of circulating androgen excess. Moreover, we demonstrate for the first time that treatment with flutamide (anti-androgen drug) is able to normalize blood cell count and haematocrit, before of achieving the definitive cure of hyperandrogenism by oophorectomy.

【 授权许可】

   
2013 Saraceno et al.; licensee BioMed Central Ltd.

【 图 表 】

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【 参考文献 】

• [1]Sarfati J, Bachelot A, Coussieu C, et al.: Study Group Hyperandrogenism in Postmenopausal Women. Impact of clinical, hormonal, radiological, and immunohistochemical studies on the diagnosis of postmenopausal hyperandrogenism. Eur J Endocrinol 2011, 165:779-788.
• [2]Pugeat M, Mirakian P, Dutrieux-Berger N, Forest MG, Tourniaire J: Androgen secreting ovarian neoplasm. In Androgen Excess Disorders in Women. Edited by Azziz R, Nestler JE, Dewailly D. Philadelphia: Lippincott-Raven; 1997:555-568.
• [3]Stephens JW, Katz JR, McDermott N, et al.: An unusual steroid-producing ovarian tumour: case report. Hum Reprod 2002, 17:1468-1471.
• [4]Yetkin DO, Demirsoy ET, Kadioglu P: Pure leydig cell tumour of the ovary in a post-menopausal patient with severe hyperandrogenism and erythrocytosis. Gynecol Endocrinol 2011, 27:237-240.
• [5]Bohlmann MK, Rabe T, Sinn HP, et al.: Intraoperative venous blood sampling to localize a small androgen-producing ovarian tumor. Gynecol Endocrinol 2005, 21:138-141.
• [6]Reman O, Reznik Y, Casadevall N, et al.: Polycythemia and steroid overproduction in a gonadotropin-secreting seminoma of the testis. Cancer 1991, 68:2224-2229.
• [7]Stephen MR, Lindop GB: A renin secreting ovarian steroid cell tumour associated with secondary polycythaemia. J Clin Pathol 1998, 51:75-77.
• [8]Girsh T, Lamb MP, Rollason TP, Brown LJ: An endometrioid tumour of the ovary presenting with hyperandrogenism, secondary polycythaemia and hypertension. BJOG 2001, 108:330-332.
• [9]McCarthy-Keith DM, Hill M, Norian JM, et al.: Use of F 18-fluoro-D-glucose-positron emission tomography-computed tomography to localize a hilar cell tumor of the ovary. Fertil Steril 2010, 94:753.e11-753.e14.
• [10]Prassopoulos V, Laspas F, Vlachou F, et al.: Leydig cell tumour of the ovary localised with positron emission tomography/computed tomography. Gynecol Endocrinol 2011, 27:837-839.
• [11]Picón MJ, Lara JI, Sarasa JL, et al.: Use of a long-acting gonadotrophin-releasing hormone analogue in a postmenopausal woman with hyperandrogenism due to a hilus cell tumour. Eur J Endocrinol 2000, 142:619-622.
• [12]Vollaard ES, van Beek AP, Verburg FA, et al.: Gonadotropin-releasing hormone agonist treatment in postmenopausal women with hyperandrogenism of ovarian origin. J Clin Endocrinol Metab 2011, 96:1197-1201.
• [13]Niemeier LA, Dabbs DJ, Beriwal S, Striebel JM, Bhargava R: Androgen receptor in breast cancer: expression in estrogen receptor-positive tumors and in estrogen receptor-negative tumors with apocrine differentiation. Mod Pathol 2010, 23:205-212.
• [14]Micello D, Marando A, Sahnane N, et al.: Androgen receptor is frequently expressed in HER2-positive, ER/PR-negative breast cancers. Virchows Arch 2010, 457:467-476.
• [15]Secreto G, Zumoff B: Role of androgen excess in the development of estrogen receptor-positive and estrogen receptor-negative breast cancer. Anticancer Res 2012, 32:3223-3228.
• [16]Miki Y, Suzuki T, Sasano H: Intracrinology of sex steroids in ductal carcinoma in situ (DCIS) of human breast: comparison to invasive ductal carcinoma (IDC) and non-neoplastic breast. J Steroid Biochem Mol Biol 2009, 114:68-71.
• [17]Pelusi C, Forlani G, Zanotti L, Gambineri A, Pasquali R: No metabolic impact of surgical normalization of hyperandrogenism in postmenopausal women with ovarian androgen-secreting tumours. Clin Endocrinol (Oxf) 2013, 78:533-538.
• [18]Orio F Jr, Palomba S, Cascella T, Di Biase S, Manguso F, Tauchmanovà L, Nardo LG, Labella D, Savastano S, Russo T, Zullo F, Colao A, Lombardi G: The increase of leukocytes as a new putative marker of low-grade chronic inflammation and early cardiovascular risk in polycystic ovary syndrome. J Clin Endocrinol Metab 2005, 90:2-5.
• [19]Brahm J, Brahm M, Segovia R, Latorre R, Zapata R, Poniachik J, Buckel E, Contreras L: Acute and fulminant hepatitis induced by flutamide: case series report and review of the literature. Ann Hepatol 2011, 10:93-98.