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A Spontaneous and Severe Hyperstimulation of the Ovaries Revealing a Gonadotroph Adenoma¹

Service d’Endocrinologie, Hopital Saint-Antoine, Assistance Publique-Hôpitaux de Paris (S.C.-M., P.B.); Laboratoire de Biochimie Médicale, Hopital Pitié-Salpétrière, Assistance Publique-Hôpitaux de Paris and Endocrinologie Cellulaire et Moléculaire de la Reproduction Centre National de la Recherche Scientifique Unité de Recherche Associée 1449, Université Paris VI (M.-L.K.); INSERM U-342, Hopital Saint-Vincent-de-Paul (N.L.); and Unité INSERM U-344 Hopital Necker Enfant-Malades (P.T.), Paris; and Service de Gynécologie Obstétrique, Hopital A Béclère (C.R.-B., R.F.), Clamart, France

Address all correspondence and requests for reprints to: Dr. Sophie Christin-Maitre, Service d’Endocrinologie, Hopital Saint-Antoine, 184 rue du Faubourg Saint-Antoine, 75571 Paris Cedex 12, France.

We report an unusual case of a gonadotroph adenoma in a 34-yr-old woman, revealed by a dramatic rise in the plasma estradiol (E₂) concentration (26,800 pmol/L; normal, <370), with nonsuppressed FSH and LH levels (4.9 and 2.4 mIU/mL, respectively). The PRL level was 503 ng/mL. The testosterone and progesterone levels were 7 and 17 nmol/L, respectively. The levels of inhibin , inhibin A, and inhibin B were increased compared to normal values in both the follicular (fp) and luteal (lp) phases of the menstrual cycle [inhibin , 1986 IU/L (fp normal, <700; lp normal, <1650); inhibin A, 254 pg/mL (fp normal, <20; lp normal, <120); inhibin B, 246 pg/mL (fp normal, <150; lp normal, <30 lp)]. Pituitary magnetic resonance imaging revealed a huge pituitary adenoma. After transphenoidal surgery, the patient presented with pituitary insufficiency and diabetes insipidus. RT-PCR of the tumor tissue was positive for LHß, FSHß, -subunit, and PRL. This case is of particular interest because 1) although the E₂ level was extremely high, the patient did not present with ascitis, suggesting that chronic elevated E₂ does not play a crucial role in the hyperstimulation symptoms; 2) the extreme rise in E₂ was related to the cosecretion of FSH and LH, confirming the two-cell two-gonadotropin theory; and 3) the rise in inhibin B is associated with FSH secretion, whereas the rise in inhibin A is probably due to luteinization.

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