Case report: Birth achieved after effective ovarian stimulation combined with dexamethasone in a patient with resistant ovary syndrome

Abstract

Background: Resistant ovary syndrome (ROS) is a rare endocrine disorder and there have been few reports of live births by affected patients. As gonadotropin resistance leads immature oocytes, some researchers reported few live births with in vitro maturation (IVM) of oocytes, but IVM is not always successful in ROS patients. Here, we report an original case of ROS, associated with Ig-FSHR in the serum, who achieved a live birth following ovarian stimulation combined with dexamethasone treatment.

Case presentation: The 30-year-old woman presented with secondary amenorrhea and infertility. Her serum FSH levels were found to be higher than normal, but in discordance with a normal anti-Müllerian hormone (AMH) level and antral follicle count. Genetic investigation found no mutations potentially affecting FSHR. With reference of previous ROS studies, the patient's serum was analyzed for antibodies directed against FSHR and dot blot analysis showed strong reactivity with FSHR. Then, dexamethasone was proposed to the patient, and she successfully became pregnant, finally delivering a healthy girl by caesarean section.

Conclusion: To our best knowledge, this is the first report of the successful treatment of ROS using ovarian stimulation combined with dexamethasone. In some cases of ROS, high doses of exogenous gonadotropins in combination with immunosuppressive therapy could be an effective approach.

Keywords: Case report; Dexamethasone; Immunosuppressant; Infertility; Resistant ovary syndrome.

Fig. 1

Transvaginal ultrasound scans of the bilateral ovaries. The size of the left and right ovaries was 2.7 × 1.6 cm and 3.2 × 1.4 cm, respectively. The number of antral follicles in the left and right ovary was 12 and 16, respectively, in line with the normal AMH level but in contrast with the high serum FSH and LH levels

Fig. 2

Diagram of two ovarian hyperstimulation cycles. GnRH, gonadotropin-releasing hormone; HCG, human chorionic gonadotropin; HMG, human menopausal gonadotropin; FSH, follicle-stimulating hormone; LH, luteinizing hormone; E2, estradiol; P, progesterone

Fig. 3

Immunoprecipitation followed by western blotting and dot blot analysis of anti-FSHR autoantibodies in the serum of the patient. A polyclonal antibody against FSHR (Abcam, ab113421) in TBST buffer (containing 3% BSA) was used as positive control serum with anti-FSHR autoantibodies, a healthy person’s serum was used as negative control, and sera from the patient before and after the dexamethasone treatment were used to immunoprecipitate recombinant FSHR protein purified from E. coli (ImmunoClone, IC8974-A). Samples comprising 0.6 μg of FSHR protein per well were separated on 10% SDS-PAGE gels and then transferred to nitrocellulose membranes. For the dot blot assay, FSHR protein (100 ng/dot) was applied onto nitrocellulose membranes. The membranes were incubated overnight at 4 °C with serum from the patient and the healthy control, followed by incubation with a horseradish peroxidase (HRP)-conjugated anti-human secondary antibody (Proteintech, SA00001–11) or anti-rabbit secondary antibody (Proteintech, SA00001–2). Stained bands or dots were visualized using Omni-ECL reagent (EpiZyme, SQ201). PC: positive control; BD: before the dexamethasone treatment; AD: after the dexamethasone treatment. NC: negative control; FSHR: follicle-stimulating hormone receptor