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Testicular “Hyperstimulation” Syndrome: A Case of Functional Gonadotropinoma

DOI: 10.1155/2014/194716

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Abstract:

Gonadotropins secreting pituitary tumors tend to present as sellar mass with hypogonadism. Biologically active LH secretion by these tumors resulting in elevated testosterone is extremely rare. We report a case of a 48-year-old male patient who presented with giant pituitary tumor, elevated testosterone, and elevated levels of gonadotropins. Surgical resection of the tumor resulted in normalization of gonadotropins and fall in serum testosterone to subnormal levels in the postoperative period confirming that the tumor was secreting bioactive luteinizing hormone (LH). 1. Introduction Most of the clinically nonfunctioning pituitary tumors are gonadotropin secreting tumors [1]. The initial manifestations of these gonadotroph tumors are visual defects, headache, and associated symptoms of anterior pituitary hormone deficiency [1]. Gonadotroph pituitary adenomas are inefficient producers and secretors of gonadotroph hormones: luteinizing hormone (LH), follicle-stimulating hormone (FSH), and the α-subunit of pituitary glycoprotein hormones [1]. Biologically active LH secretion by these tumors resulting in elevated testosterone is extremely rare. We report a case of a 48-year-old male patient who presented with giant pituitary tumor, elevated testosterone, and elevated levels of gonadotropins. 2. Case Summary A 48-year-old man was referred for further management of a large pituitary tumor. His wife reported his having an increased libido for the preceding 12 months. Visual examination showed a mydriatic right pupil with a diminished light response and a right temporal field defect. There was no gynecomastia. Genital examination revealed testicular volumes approximating 25–30?cc. Biochemical evaluation showed an elevated total testosterone level of 1647?ng/dL (normal 220–1000; chemiluminescence immunoassay) and free testosterone of 515.1?pg/mL (normal 40–240; chemiluminescence immunoassay and ultrafiltration), with corresponding follicular-stimulating hormone (FSH) and LH levels of 32.7?mU/mL (normal 1–10) and 11.5?mU/mL (normal 1–7), respectively, consistent with a functional gonadotropinoma. There was no other hormonal cosecretion (Table 1). Magnetic resonance imaging (MRI) of the sella revealed a 4.5 × 3.3 × 2.5?cm sellar and suprasellar tumor, with optic nerve and chiasm compression and right cavernous sinus invasion (Figure 1). The patient underwent transsphenoidal surgery with dramatic reductions in postoperative total and free testosterone levels to 128 and 18.5, along with normalization of FSH and LH levels (6.0?mU/mL and 1.5?mU/mL), respectively.

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