Ovarian granulosa cell tumor and clomiphene citrate resistance. A case report and review of the literature

Granulosa cell tumors (GCTs) account for less than 5% of all ovarian malignancies, occur in younger ages, are usually diagnosed in their early stages, and have a good prognosis. GCTs usually present with features of hyperestrogenism. This paper reports the unusual case of an adult with a GCT with manifestations including amenorrhea, mild hirsutism, infertility, clomiphene citrate (CC) resistance (CC), mildly elevated testosterone, high anti-Mullerian hormone (AMH) and normal estrogen levels. The patient was initially diagnosed with polycystic ovarian syndrome (PCOS), and after four attempts at ovarian stimulation she was diagnosed with CC resistance. The patient later underwent laparoscopic evaluation on account of a solid mass on her left ovary. The pathology report described it as a borderline adult type GCT and four weeks after surgery she had a positive pregnancy test. Twelve months after delivery, the patient had no obvious symptoms of disease and her menstrual cycle was normal. Serial measurements of serum inhibin B, AMH, estrogen, and testosterone levels were within normal range. In conclusion, the resistance to clomiphene manifested by the patient might be explained by a potential mechanism implicating inhibin B and AMH due to the presence of a GCT. Further studies are required to evaluate the role of AMH and Inhibin B in the mechanism of CC resistance in women with PCOS.