Abstract
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.
Highlights
Severe female hyperandrogenism is an endocrine disorder characterized by serum testosterone levels in the masculine range
In this paper we report for the first time the association of Leydig cell tumour and androgen receptors (AR)+ve breast cancer in a woman with a long-term history of infertility, dramatic virilism and erythrocytosis
The finding that the breast tumour did not express ER or HER2 suggests that cell proliferation can be stimulated by androgen excess, independently by circulating estrogens concentration if AR are expressed, and is in keeping with the androgen-excess theory recently proposed by Secreto and Zumoff [15]
Summary
Severe female hyperandrogenism is an endocrine disorder characterized by serum testosterone levels in the masculine range. This is a very rare condition which is caused by adrenal or ovarian dysfunction, genetic or proliferative diseases. We present the case of a middle-age woman affected by an occult androgen secreting tumour of ovary causing erythrocytosis and by a breast cancer expressing only androgen receptors (AR) and, at lower extent, progesterone receptors (PR). The association of these two neoplasms has not been reported until now and. We will speculate on the proliferative role of severe hyperandrogenism on clusters of AR positive (AR+ve) breast tumour cells
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