Spontaneous ovarian hyperstimulation syndrome and pituitary adenoma: incidental pregnancy triggers a catastrophic event

Objective To report a rare case of spontaneous ovarian hyperstimulation syndrome (OHSS) associated with spontaneous pregnancy and a FSH-secreting pituitary adenoma. Design Case report. Setting University hospital. Patient(s) A 32-year-old woman with spontaneous OHSS. Intervention(s) Transsphenoidal resection of the tumor. Main Outcome Measure(s) Regression of the symptoms of OHSS and hyperestrogenemia. Result(s) At presentation, the patient's ovaries were markedly enlarged and massive ascites was seen. Her serum E 2 level was markedly elevated, but her LH level was low, and FSH was within the normal range. In addition, her TSH level was normal, and hCG was appropriate for the date of pregnancy. Subsequently, the patient developed massive thrombophlebitis in her right internal jugular and subclavian veins. Termination of the pregnancy ameliorated the accumulation of ascites, but ovarian enlargement and hyperestrogenemia persisted. No mutations of the FSH receptor, LH receptor, or aromatase genes were detected, but magnetic resonance imaging (MRI) of the head revealed a pituitary adenoma. After transsphenoidal resection of the tumor, the patient got better. Conclusion(s) A gonadotropin-secreting adenoma caused ovarian hyperstimulation (ovarian enlargement and hyperestrogenemia). In addition, spontaneous pregnancy and intrinsic hCG increased vascular permeability, which complicated the patient's disease.