Successful Pregnancy after Bromocriptine Therapy in an Anovulatory Woman Complicated with Ovarian Hyperstimulation Caused by Follicle-Stimulating Hormone-Producing Plurihormonal Pituitary Microadenoma

Abstract

Gonadotropin-producing pituitary adenomas are extremely rare in reproductive-age women. We report here a case of gonadotroph microadenoma with ovarian hyperstimulation. It was found in a 29-yr-old infertile Japanese woman with enlarged multicystic ovaries. The patient had an elevated basal serum estradiol level (up to 6755 pM, or 1840 pg/ml). Serum FSH and prolactin were mildly elevated (15.4 IU/liter, 1.4 nM or 31.4 ng/ml), whereas LH was low (0.5 IU/liter). The FSH level was paradoxically elevated in response to TRH administration. Dynamic magnetic resonance imaging revealed a pituitary microadenoma. Daily administration of bromocriptine, a dopamine agonist, normalized the ovarian size, and the patient ovulated naturally. She conceived after 3 months of bromocriptine therapy and delivered a normal child. She underwent elective transsphenoidal pituitary surgery, 3 yr after the delivery. Immunostaining of the resected tumor showed that 80% and less than 5% of the tumor cells stained for FSH-beta and prolactin, respectively. Furthermore, RT-PCR suggested that dopamine type 2 receptor was expressed in the adenoma. Gonadotroph microadenoma should be considered in women with spontaneous ovarian hyperstimulation, even if they have no neurological symptoms or marked pituitary enlargement. In such cases, bromocriptine therapy may be an alternative to pituitary surgery.

A Case of Acquired Deficiency of Pituitary GH, PRL and TSH, Associated with Type 1 Diabetes Mellitus

“…Dizon and Vesely reported a 61-year-old male who showed high plasma levels of FSH, LH, PRL and total T [10]. In addition, some reports showed female cases who showed high levels of FSH and estradiol [11][12][13]. Moreover, Shimon et al reported a case of a 28-year-old female with a high concentration of FSH and a normal level of estradiol [14].…”

Ovarian hyperstimulation syndrome caused by an FSH-secreting pituitary adenoma

“…In theory, one possible medical treatment would be the administration of a GnRH analog which would decrease FSH levels, thereby leading to resolution of OHSS; however, reports have actually shown a paradoxical increase in gonadotropin secretion in response to this treatment, 4,[15][16][17][18][19][20] and in one case OHSS was induced after initiation of GnRH therapy with dramatic increases in FSH and estradiol levels. 21 There are three reports of patients with OHSS who were initially treated medically.…”

“…Eventually, however, the adenomas continued to grow and were resected, showing positive immunostaining for LH in two cases and for FSH in one case. 15,22 After resection of a pituitary adenoma, patients are monitored annually with MRI, looking for evidence of a possible recurrence. Patients should also undergo hormonal testing 3 months after surgery to assess whether hypo-pituitarism is present.…”

Effect of temozolomide on cell viability in gonadotroph adenoma cell lines

“…Baseline concentrations of gonadotropin are elevated in 30-50% of gonadotroph adenoma patients, and may result in ovarian hyperstimulation and dysgenesia (28)(29)(30)(31). In previous clinical treatment of gonadotroph adenoma patients with TMZ, changes in gonadotropin levels before and after drug administration were not evaluated (15,16).…”