Ovarian hyperstimulation syndrome
Rarely, gonadotroph adenomas induce an ovarian hyperstimulation syndrome in females. Ovarian hyperstimulation could regress after resolving the causes of high follicle-stimulating hormone level, so avoiding unnecessary ovary surgery. Detailed endocrinological examination including estradiol evaluation with pituitary imaging is quite important in women of reproductive age to establish the correct diagnosis 1) 2).
Ovarian hyperstimulation syndrome (OHSS) is an iatrogenic disorder associated with infertility treatment. The main pathology of OHSS is intravascular dehydration and hyperestrogenism. In mild cases, abdominal symptoms are the main symptoms, but in severe cases, thrombosis such as cerebral infarction may occur.
A 36-year-old woman was undergoing infertility treatment in obstetrics and gynecology for infertility. She received HMG-HCG therapy and artificial insemination one week before onset, and on the day of onset she had mild abdominal distension and was suspected of OHSS. She was prescribed aspirin for the prevention of thrombosis. She presented with right upper hemiparesis and aphasia. MRI showed left middle cerebral artery occlusion. They performed mechanical thrombectomy and finally got Thrombolysis in Cerebral Infarction scale (TICI) 3 recanalization.
Kuwano et al. suspected embolism as the etiology of cerebral infarction and started anticoagulation therapy. Various examinations were conducted to investigate the embolism source, but no anatomical abnormality or thrombophilia factors were observed. The patient had admitted OHSS since admission, we concluded that OHSS was suspected as the cause of this stroke.
Kuwano et al. experienced the first case of mechanical thrombectomy for middle cerebral artery occlusion suspected to be caused by OHSS. It is necessary to suspect OHSS involvement if young women, especially those on infertility treatment, show neurological deficits 3).
Graillon et al. reported 2 cases of functioning gonadotroph pituitary neuroendocrine tumor (FGPA) revealed by an ovarian hyperstimulation syndrome (OHSS) in young women. In the first case, OHSS was observed after GnRH analog injection. Pelvic echography revealed multiple voluminous ovarian cysts. Dopamine agonist posology failed in estradiol hypersecretion control, which necessitated endoscopic endonasal transsphenoidal surgery. The patient experienced improvement in pelvic pain as estradiol hypersecretion decreased during the first few postoperative days. Outcome was favorable, and her menstrual cycle was normal after two months. The second case was a young girl with spontaneous pelvic pain and elevated plasma FSH and estradiol levels. FGPA was confirmed on cerebral MRI. Dopamine agonists were introduced, and surgical removal of the pituitary tumor was scheduled for 7 days later. In the meantime, the patient was admitted and underwent surgery for bilateral adnexal torsion related to OHSS. The pituitary tumor was removed one week later. Outcome was favorable, and estradiol and FSH plasma levels were normal after 3 months. The ovarian cysts were no longer visible on echography after 3 months. Given the lack of efficacy of the current standard medical therapy, surgical removal of pituitary neuroendocrine tumors is the reference treatment for FGPA. The authors suggest that severe OHSS related to FGPA should be considered as a relative surgical emergency and that surgery should not be unduly delayed, given the unpredictable risk of adnexal torsion, particularly in case of voluminous ovarian cysts. The authors performed a literature review on this topic 4).
Azriel et al. presented a rare patient with ascites following ovarian hyperstimulation syndrome (OHSS) leading to shunt malfunction. OHSS is a potentially life-threatening complication of controlled ovarian stimulation caused by the administration of exogenous gonadotropins. In this patient clinical and radiological resolution of shunt dysfunction was achieved following peritoneocentesis. To our knowledge this is the first described case of OHSS leading to shunt malfunction, emphasizing the importance of awareness, early recognition and proper management of abdominal etiologies of VPS malfunction 5).
Ovarian hyperstimulation from ectopic hypersecretion of follicle stimulating hormone 6).
In 2014 Kumako et al. reported the cases of three young women aged 35, 37 and 27 years. All three were victims of ischemic formed by proximal occlusion of the middle cerebral artery secondary to ovarian hyperstimulation. The first and the third had a proximal occlusion of the right middle cerebral artery occlusion and the second of the left middle cerebral artery. The last two have benefited from a patient intravenous thrombolysis. The first patient did not receive thrombolysis because it was out of time. Against by their evolution was different. The first has almost recovered its deficit, the second sequelae quite heavy after craniectomy and the third died despite her craniectomy 7).
Lee et al. reported on a patient who presented with shunt failure due to ovarian hyperstimulation syndrome (OHSS) following in vitro fertilization treatment. Shunt dysfunction was attributed to intraabdominal hypertension as a consequence of ascites. At surgery, the shunt was found to be patent. The peritoneal catheter was externalized and subsequently revised to become a ventriculoatrial shunt system. This led to clinical improvement in the patient and the restoration of ventricular size. Such a shunt complication has not previously been reported. Neurosurgeons should be alerted to this possibility in view of the increasing use of assisted conception in many developed countries 8).