| Summary: | Background: Although the incidence is rare, patients with moyamoya disease are at risk of stroke during pregnancy. Therefore, there is limited information regarding appropriate peripartum management. Herein, we report a case of major postpartum cerebral infarction in a patient with moyamoya disease, despite previous direct bilateral anastomosis of the superficial temporal artery to the middle cerebral artery (STA-MCA). Case description: The patient was a 32-year-old woman who had undergone direct STA-MCA anastomosis and encephalo-myo-synangiosis in her childhood. This was her second pregnancy. She sustained a major ischemic stroke in the left middle cerebral artery area on postpartum day 3, which resulted in right hemiplegia and aphasia. On magnetic resonance angiography, simultaneous interruption of antegrade flow of the left middle cerebral artery and STA-MCA anastomosis was observed. The patency of these vessels was confirmed before the first pregnancy. The patient’s consciousness was impaired for several days due to edema in the infarct area. However, her condition improved following conservative treatment with osmotic diuretics and antiplatelet therapy; the patient recovered independent gait and her aphasia improved after 5 months of inpatient rehabilitation. Conclusions: Careful monitoring of women with moyamoya disease is recommended during pregnancy and the peripartum period even if direct or indirect anastomosis has been previously performed.
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