Pseudo‐isolated FSH deficiency caused by an inhibin B‐secreting granulosa cell tumour: Case report

• Published in: Human reproduction (Oxford) Vol. 18; no. 3; pp. 502 - 505
• Main Authors: Krishnan, A., Murdock, C., Allard, J., Cisar, M., Reid, E., Nieman, L., Segars, J.
• Format: Journal Article
• Language: English
• Published: Oxford Oxford University Press 01.03.2003; Oxford Publishing Limited (England)
• Subjects: Adult; Biological and medical sciences; Delivery, Obstetric; Female; Female genital diseases; Fertilization; Follicle Stimulating Hormone - deficiency; Granulosa Cell Tumor - complications; Granulosa Cell Tumor - diagnostic imaging; Granulosa Cell Tumor - metabolism; Granulosa Cell Tumor - surgery; Gynecology. Andrology. Obstetrics; Humans; Immunohistochemistry - methods; Infertility, Female - etiology; Inhibins - metabolism; Key words: FSH deficiency/granulosa cell tumour/hypothalamic amenorrhoea/infertility/inhibin B; Medical sciences; Ovarian Neoplasms - complications; Ovarian Neoplasms - diagnostic imaging; Ovarian Neoplasms - metabolism; Ovarian Neoplasms - surgery; Recovery of Function; Staining and Labeling; Tumors; Ultrasonography; FSH deficiency/granulosa cell tumour/hypothalamic amenorrhoea/infertility/inhibin B; Human; Menstruation disorders; Deficiency; Inhibin; Female sterility; Gonadotropin; Female genital diseases; Case study; Ovarian diseases; Ovary; Adenohypophyseal hormone; Female genital system; Amenorrhea; Female; Tumor; Granulosa; Exocrine secretion; Ovarian follicle; Tumor cell; Follicle stimulating hormone
• ISSN: 0268-1161; 1460-2350; 1460-2350
• DOI: 10.1093/humrep/deg124

Isolated FSH deficiency due to a mutation in the FSHβ subunit is characterized by an extremely low serum FSH concentration. We report a patient who presented with an FSH of 0.8 mIU/ml and infertility associated with anovulation. Endocrinological assessment and immunohistochemistry revealed that a granulosa cell tumour was secreting inhibin B and suppressing FSH; however, LH and estradiol were within their normal ranges. Upon removal of the tumour, inhibin B decreased and FSH levels rose to normal values. The patient subsequently conceived and delivered successfully. Based on this case and on those previously described in the literature, we suggest that inhibin B levels should be evaluated in anovulatory patients having a clinical presentation consistent with functional hypothalamic amenorrhoea and very low to normal values of FSH.