CASE REPORT: Management of ovarian hyperstimulation due to follicle‐stimulating hormone‐secreting gonadotroph adenoma

A 21 year old virgin woman sought medical attention because of a history of secondary amenorrhoea for several years and abdominal distention for the previous six months. She had been receiving a cyclic oestrogen and progestin therapy, and bilateral ovarian cysts had been detected at previous clinics that she had visited. Pelvic ultrasonography and magnetic resonance imaging (MRI) showed bilateral multiple ovarian cysts over the pelvis, a small volume of ascites and a relatively thickened endometrium (Fig. 1). There was no solid portion in the cysts. The serum concentrations of tumour markers were not elevated and were as follows: CA125, 38.6 U/mL (normal 39 U/mL); CA19–9, 27.7 U/mL ( 37 U/mL); carcinoembryonic antigen, 1.43 ng/mL ( 5 ng/mL); and a-fetoprotein, 0.80 ng/mL ( 20 ng/mL). Endocrinological examination showed that the serum oestradiol concentration was extremely high at 3998 pmol/L. The serum follicle-stimulating hormone (FSH) and prolactin (PRL) concentrations were supranormal at 10.3 IU/mL and 79.9 ng/L, respectively. Serum luteinising hormone (LH) was not detectable before and even after administration of LH-releasing hormone or thyrotropin-releasing hormone. Haemoconcentration and electrolyte abnormalities were not seen. Terguride therapy was administered and a brain MRI was planned due to mild hyperprolactinaemia and galactorrhoea. The MRI revealed a pituitary tumour. Visual field examination, which was performed after the MRI, revealed bitemporal hemianopsia. The levels of other pituitary hormones were within the normal ranges and were as follows: growth hormone (GH), 1.68 ng/mL; thyrotropinstimulating hormone (TSH), 1.05 AIU/mL; and adenocorticotropic hormone (ACTH), 36.7 pg/mL. Terguride therapy was cancelled in a week, and transsphenoidal removal of the pituitary adenoma was performed as the initial therapy. Her ovarian cysts were not treated because she did not have severe abdominal pain and there was a low possibility of malignancy. Immunohistochemical staining of the pituitary tumour for pituitary hormones revealed that the tumour cells expressed LH, FSH and PRL but not GH, TSH or ACTH. The patient was diagnosed with gonadotroph adenoma. Her post-operative course was uneventful. She had withdrawal bleeding for several days after the operation. The bilateral huge ovarian cysts dramatically decreased in size and her hormone levels of FSH, PRL, oestradiol and LH improved within one month after the operation. Other pituitary hormone levels were not influenced by the transsphenoidal surgery. Her menstrual cycle resumed, and her visual field deficits improved gradually. At 26 years of age, five years after excision of the pituitary tumour, she had no recurrent signs of gonadotroph adenoma. She conceived spontaneously during the following year. She delivered a female baby weighing 2488 g by spontaneous vaginal delivery at 37 weeks of gestation.