Ovarian Hyperstimulation Syndrome Caused by an FSH-Secreting Pituitary Adenoma

Odelia Cooper; Jordan L Geller; Shlomo Melmed

Disclosures

Nat Clin Pract Endocrinol Metab. 2008;4(4):234-238. 

In This Article

The Case

A 40-year-old woman presented in 2006 with a 15-year history of bilateral galactorrhea. She has never been pregnant and had initially presented at the age of 25 years with menometrorraghia (dysfunctional uterine bleeding) and bilateral galactorrhea, while being on oral contraceptives. At that time investigations revealed the presence of benign luteinized follicular cysts, which were resected laparoscopically. Her symptoms then temporarily abated, but recurred with re-growth of ovarian cysts and with features of the ovarian hyperstimulation syndrome (OHSS) including pelvic pain and enlarged, multicystic ovaries, which required three separate operations over the following 5 years.

The patient was referred for endocrinological evaluation in 2006, because she had persistent galactorrhea. At this time, she reported having had a normal adrenarche and puberty, and she denied suffering from headaches, visual disturbances, weight or body habitus changes, polyuria, hirsutism or acne. Her history revealed that she was receiving the oral contraceptive pill. On physical examination, she was found to have bilateral galactorrhea.

Biochemical testing demonstrated a negative pregnancy test, prolactin levels of 3,000 pmol/l (reference range 165-1,009 pmol/l), follicle-stimulating hormone (FSH) levels of 19.2 IU/l (reference range 4-13 IU/l), luteinizing hormone (LH) levels of 0.6 IU/l (reference range 1-18 IU/l), estradiol levels of 3,851.0 pmol/l (reference value <1,094.0 pmol/l), adrenocorticotropic hormone levels of 2.9 pmol/l (reference range 1.1-5.9 pmol/l), 0800 h cortisol levels of 510.4 nmol/l (reference range 165.5-634.6 nmol/l), glycoprotein hormone α subunit levels of 0.8 µg/l (reference range 0.04-0.38 µg/l), TSH levels of 3.3 mIU/l (reference range 0.39-4.6 mIU/l), free endogenous T4 levels of 11.6 pmol/l (reference range 10.3-34.7 pmol/l), and insulin-like growth factor I levels of 33.7 nmol/l (reference range 18.1-53.7 nmol/l). MRI of the pituitary gland revealed a 16 x 27 x 22 mm pituitary adenoma with suprasellar extension, elevation and compression of the optic chiasm, and extension to the right cavernous sinus (Figure 1). The patient underwent trans-sphenoidal resection of the pituitary mass.

The patient's pre-operative MRI depicting the pituitary mass. (A) Coronal view and (B) sagittal view.

Immunohistochemical staining of the pituitary adenoma specimen was positive for α subunit, FSHß subunit and LHß subunit; staining was negative for growth hormone, prolactin, adrenocorticotropic hormone and for TSH. The Ki-67 proliferative index, measuring the growth fraction of tumor cells, was low (1%) and almost no cells showing overexpression of p53 protein were detected. These findings generally suggests a low tumor grade and a good prognosis.

The patient was diagnosed as harboring a gonadotrope cell adenoma with secondary ovarian hyperstimulation. Postoperatively, galactorrhea resolved, and normal menses resumed. Anterior pituitary hormone reserve was intact.

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