Evolution of clinical symptoms in a young woman with a recurrent gonadotroph adenoma causing ovarian hyperstimulation

Abstract

Objective: To demonstrate the clinical course in a young female with gonadotroph adenoma causing ovarian stimulation. Patient and methods: Our patient was a 23-year-old woman with a history of oligomenorrhea who had previously undergone bilateral ovarian wedge resection owing to the clinical appearance of polycystic ovaries. Two years later, she sought treatment for headache, galactorrhea, history of spotting and lower abdominal distension. FSH, LH, b-LH, inhibin A and B, estradiol, prolactin (PRL), and bchorionic gonadotrophin (b-CG) were measured, and the responses of FSH, LH and b-LH to thyrotrophin-releasing hormone (TRH) were documented. Immunohistochemical analysis of the tumor tissue was performed after surgery. Five years after the trans-sphenoidal surgery, the patient again became oligomenorrheic. A large recurrent adenoma was diagnosed on CT one year later. Transvaginal ultrasound showed ovaries of normal size with multiple small cystic formations simulating a polycystic pattern, While the patient was awaiting surgery, a pituitary apoplexy occurred. Emergency decompressive surgery was performed and the patient fully recovered. Results: Enlarged ovaries were found on ultrasound examination simulating a hyperstimulationlike pattern. At that time, elevated levels of FSH (13.4 IU/l) and marginally elevated levels of b-LH (1.43 ng/ml) were found, whereas the level of LH (0.5 IU/l) was subnormal. Plasma estradiol was markedly supranormal (6150 pmol/l). Levels of inhibin A and B were elevated (326 pg/ml and 588 pg/ml respectively). The prolactin level (70 ng/ml) was increased, whereas b-chorionic gonadotrophin (b-CG) was normal. Signi®cantly increased FSH, LH, and b-LH responses to TRH stimulation were documented. Pituitary macroadenoma was found on MRI scan and removed by trans-sphenoidal surgery. Immunohistochemical examination showed high positivity for b-CG and LH, and slight positivity for FSH. Five years after the surgery, estradiol was elevated (1160 pmol/l), whereas basal levels of LH (4.65 IU/l) and FSH (3.98 IU/l) were not suppressed. After the second operation, immunostaining of the adenoma tissue con®rmed the previous ®ndings. Conclusions: Measurement of gonadotrophins in our case did not prove to be a method for identifying a large recurrent gonadotroph pituitary adenoma. The sonographic ovarian imaging varied from a polycystic-to an ovarian hyperstimulation-like pattern during the evolution of the tumour.

Ovarian hyperstimulation syndrome caused by an FSH-secreting pituitary adenoma

“…2,3 Women with intact gonadotrope adenomas and supranormal FSH levels are generally not recognized as exhibiting a syndrome, because many are over 45 years of age with ovaries devoid of pre-antral follicles, and are insensitive to the action of FSH. 4 These gonadotrope adenomas are, therefore, more difficult to diagnose in women who are perimenopausal or postmenopausal. 5 In women, gonadotropinomas should also be considered if there is a history of headaches or visual changes.…”

“…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.…”

Ovarian Hyperstimulation Caused by Gonadotroph Pituitary Adenoma – Review

“…Elevated α-subunit levels may indicate the presence of pituitary adenoma [8,9]. FSH secreted by the tumor cells causes the formation of the multiple ovarian cysts and may considerably increase the production of estradiol [21,22]. In some reports high estradiol levels contributed to the development of endometrial hyperplasia [17,21,23].…”

Follicle stimulating hormone secreting pituitary adenoma: a challenging diagnosis

“…To determine sure nature of pituitary tumour, other pituitary hormonal assays were done -Serum ACTH -38 pg/ml (0-46), Serum cortisol -1.3 mcg/ml (3)(4)(5)(6)(7)(8)(9)(10)(11)(12)(13)(14)(15)(16). It confirmed the nature of tumour as 'mixed pituitary adenoma' secreting FSH and prolactin.…”

“…Women with intact gonadotrope adenomas and supra-normal FSH levels are generally not recognized as exhibiting a syndrome, because many are over 45 years of age with ovaries devoid of pre-antral follicles, and are insensitive to the action of FSH. 6 These gonadotrope adenomas are, therefore, more difficult to diagnose in women who are perimenopausal or postmenopausal. There have been few reports worldwide that have revealed that serum FSH levels were normal to slightly high in patients with combined FSH -secreting pituitary adenoma with ovarian hyperstimulation.…”