S. Christinmaitre et al., A SPONTANEOUS AND SEVERE HYPERSTIMULATION OF THE OVARIES REVEALING A GONADOTROPH ADENOMA, The Journal of clinical endocrinology and metabolism, 83(10), 1998, pp. 3450-3453
We report an unusual case of a gonadotroph adenoma in a 34-yr-old woma
n, revealed by a dramatic rise in the plasma estradiol (E-2) concentra
tion (26,800 pmol/L; normal, <370), with nonsuppressed FSH and LH leve
ls (4.9 and 2.4 mIU/mL, respectively). The PRL level was,503 ng/mL. Th
e testosterone and progesterone levels were 7 and 17 nmol/L, respectiv
ely. The levels of inhibin alpha, inhibin A, and inhibin B were increa
sed compared to normal values in both the follicular (fp) and luteal (
Ip) phases of the menstrual cycle [inhibin alpha 1986 IU/L (fp normal,
<700; Ip normal, <1650); inhibin A, 254 pg/mL (fp normal, <20; Ip nor
mal, <120); inhibin B, 246 pg/mL (fp normal, <150; Ip normal, <30 Ip)]
. Pituitary magnetic resonance imaging revealed a huge pituitary adeno
ma. After transphenoidal surgery, the patient presented with pituitary
insufficiency and diabetes: insipidus. RT-PCR of the tumor tissue was
positive for LH beta, FSH beta, alpha-subunit, and PRL. This case is
of particular interest because 1) although the E-2 level was extremely
high, the patient did not present with ascitis, suggesting that chron
ic elevated E-2 does not play a crucial role in the hyperstimulation s
ymptoms; 2) the extreme rise in E-2 was related to the cosecretion of
FSH and LH, confirming the two-cell two-gonadotrophin theory; and 3) t
thee rise in inhibin B is associated with FSH secretion, whereas the r
ise in inhibin A is probably due to luteinization.