Ovarian hyperstimulation, hyperprolactinaemia and LH gonadotroph adenoma

Abstract

This report considers a highly exceptional case of ovarian hyperstimulation syndrome due to a gonadotroph adenoma secreting LH in a 31-year-old patient who presented with amenorrhoea and galactorrhoea syndrome and a complex bilateral ovarian mass. Magnetic resonance imaging revealed a pituitary adenoma, and laboratory tests corroborated the hyperprolactinaemia without other hormonal pituitary abnormalities. Ovarian hyperstimulation syndrome due to a gonadotroph adenoma with normal gonadotrophins is extremely rare. Most of the described cases are caused by FSH adenomas. Due to the originality of the case, it was considered useful for understanding the management of this entity, and it is proposed that LH adenomas should also be considered in the differential diagnosis of patients with spontaneous ovarian hyperstimulation syndrome.

Isolated luteinizing hormone (LH) elevation in a woman with secondary amenorrhea: a clue to the diagnosis of an inhibin B–producing thecoma and insights into the influence of inhibin B on LH

“…Rare reports of gonadotropin-producing pituitary adenomas have been associated with elevated E and hyperstimulation syndrome, rather than the hypoestrogenization state of our patient (7,8). Had our workup for this patient not have led so quickly to a diagnosis, suppressability of the LH to GnRH agonist could have helped to exclude the diagnosis of an LH-secreting pituitary adenoma.…”

Radiological Illustration of Spontaneous Ovarian Hyperstimulation Syndrome

“…The clinical and radiological spectrum is similar to the one of much more common, ovulation therapy-induced OHSS, which complicates 1% of assisted reproduction techniques [ 1 ]. The s-OHSS is usually seen to complicate pregnancy (both singleton and multiple) [ 2 – 8 ], gestational trophoblastic tumor [ 9 – 12 ], pituitary adenomas [ 13 – 19 ], hypothyroidism [ 20 – 23 ], and β hCG-secreting tumors. A single case of OHSS due to ectopic FSH secretion by a neuro-endocrine tumor in the thorax has also been reported [ 25 ], with no case yet reported in the literature of non-gonadal β hCG -secreting tumor causing OHSS, as in our case.…”

“…Ovarian hyperstimulation syndrome (OHSS) is a rare, potentially life-threatening systemic complication caused by iatrogenic and spontaneous (non-iatrogenic) ovarian stimulation occurring due to little-known patho-physiological mechanisms. The vast majority of OHSS cases are usually iatrogenic and are seen to complicate about 1 percent of all assisted reproductive technologies, and are induced by exogenous hormonal therapy [ 1 ], whereas the extremely rare, non-iatrogenic cases, which are also known as spontaneous OHSS (s-OHSS) are unrelated to exogenous hormones and are usually seen to complicate pregnancy (both singleton and multiple) [ 2 – 8 ], gestational trophoblastic neoplasms [ 9 – 12 ], pituitary adenomas [ 13 – 19 ], and hypothyroidism [ 20 – 23 ]. Clinicoradiological spectrum ranges from mild to severe [ 24 ] OHSS which needs prompt evaluation and management.…”

Follicle-Stimulating Hormone-Producing Pituitary Adenoma: A Case Report and Review of the Literature

“…While the majority of cases of OHSS involve FSH-secreting tumors with the hormonal profile described above, several of these have some positivity for LH on histology. There has also been a report of an LH-secreting pituitary adenoma causing OHSS, where FSH and LH levels were normal, but estradiol was elevated (15).…”

“…Estrogen is associated with prothrombotic alterations in coagulation proteins and has been commonly associated with hypercoagulation (18). Despite the very high levels of estrogen, OHSS caused by gonadotropin-secreting adenomas does not typically present with ascites or hypercoagulability for unclear reasons (15). Only 1 case report describes a patient with an FGA and estradiol levels reaching 18,617 pg/mL who presented with thromboembolism (19).…”