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Testicular "hyperstimulation" syndrome: a case of functional gonadotropinoma.

Thakkar A, Kannan S, Hamrahian A, Prayson RA, Weil RJ, Faiman C - Case Rep Endocrinol (2014)

Bottom Line: Gonadotropins secreting pituitary tumors tend to present as sellar mass with hypogonadism.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).

View Article: PubMed Central - PubMed

Affiliation: Topiwala National Medical College and BYL Nair Charitable Hospital, Mumbai 400008, India.

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).

No MeSH data available.


Related in: MedlinePlus

(a) H&E staining (magnification ×200) is characterized by many chromophobic epithelioid cells arranged in “pseudorosette” patterns. (b) Positive immunostaining of the tumor for FSH antibody and M3504 polyclonal antibody (DAKO, Carpinteria, CA) in 1 : 200 dilution. (c) Negative immunostaining for LH antibody (in (c)), M3502, and C93 clone (DAKO) in 1 : 320 dilution.
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fig2: (a) H&E staining (magnification ×200) is characterized by many chromophobic epithelioid cells arranged in “pseudorosette” patterns. (b) Positive immunostaining of the tumor for FSH antibody and M3504 polyclonal antibody (DAKO, Carpinteria, CA) in 1 : 200 dilution. (c) Negative immunostaining for LH antibody (in (c)), M3502, and C93 clone (DAKO) in 1 : 320 dilution.

Mentions: 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. Postoperative MRI showed significant tumor debulking. Immunohistochemically, the adenoma (Figure 2(a)) stained positively for FSH (Figure 2(b)) but did not stain for LH (Figure 2(c)). Staining was repeated on different specimens of the tumor and the tumor consistently did not stain for LH. Slides prepared were not conducive for electron microscopy.


Testicular "hyperstimulation" syndrome: a case of functional gonadotropinoma.

Thakkar A, Kannan S, Hamrahian A, Prayson RA, Weil RJ, Faiman C - Case Rep Endocrinol (2014)

(a) H&E staining (magnification ×200) is characterized by many chromophobic epithelioid cells arranged in “pseudorosette” patterns. (b) Positive immunostaining of the tumor for FSH antibody and M3504 polyclonal antibody (DAKO, Carpinteria, CA) in 1 : 200 dilution. (c) Negative immunostaining for LH antibody (in (c)), M3502, and C93 clone (DAKO) in 1 : 320 dilution.
© Copyright Policy
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC3926319&req=5

fig2: (a) H&E staining (magnification ×200) is characterized by many chromophobic epithelioid cells arranged in “pseudorosette” patterns. (b) Positive immunostaining of the tumor for FSH antibody and M3504 polyclonal antibody (DAKO, Carpinteria, CA) in 1 : 200 dilution. (c) Negative immunostaining for LH antibody (in (c)), M3502, and C93 clone (DAKO) in 1 : 320 dilution.
Mentions: 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. Postoperative MRI showed significant tumor debulking. Immunohistochemically, the adenoma (Figure 2(a)) stained positively for FSH (Figure 2(b)) but did not stain for LH (Figure 2(c)). Staining was repeated on different specimens of the tumor and the tumor consistently did not stain for LH. Slides prepared were not conducive for electron microscopy.

Bottom Line: Gonadotropins secreting pituitary tumors tend to present as sellar mass with hypogonadism.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).

View Article: PubMed Central - PubMed

Affiliation: Topiwala National Medical College and BYL Nair Charitable Hospital, Mumbai 400008, India.

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).

No MeSH data available.


Related in: MedlinePlus