Adenomyosis is a common, benign uterine disease. A pathological trait of the disease is the formation of uterine glands and stroma in the endometrial membrane. It presents with abnormal uterine bleeding, pelvic pain, and uterine growth in volume, with a prevalence of 20–35% in women. The exact diagnosis is based on pathological findings, but the images obtained by radiological tests and tumor markers may be sufficient to diagnose adenomyosis by radiologists or gynecologists. Trousseau's syndrome (TS) is characterized by cancer-related embolic events, such as deep vein thrombosis/pulmonary embolism and cerebral infarction (CI). Acute CIs are mainly caused by smoking, hypertension, dyslipidemia, abnormal glucose tolerance, and aging. Clinicians usually consider TS when observing patients with acute CIs of an unknown origin. TS accounts for a small number of patients among all stroke diseases as a cause, but it should not be misdiagnosed due to its fatality. To date, 16 cases of adenomyosis that resemble TS have been reported. This disease shows relatively similar radiographical and demographical presentations to gynecological cancers, which confuses patients and doctors. Additionally, no standardized treatments have been verified to date. Here, we report a successfully treated extremely rare case of adenomyosis associated with recurrent acute CI