"Recurrent multiple cerebral infarctions related to the progression of adenomyosis: a case report"

• Published in: BMC neurology 2018-08, Vol.18 (1), p.119-119, Article 119
• Main Authors: Aso, Yasuhiro, Chikazawa, Ryo, Kimura, Yuki, Kimura, Noriyuki, Matsubara, Etsuro
• Format: Article
• Language: English
• Subjects: Adenomyosis; Anticoagulants; Anticoagulation therapy; Brain infarction; Care and treatment; Case Report; Case reports; Case studies; Causes of; Cerebellum; Cerebral infarction; Cerebrum; Complications and side effects; Cortex; Echocardiography; Endometriosis; Gait; Headache; Heparin; Hypercoagulagulability; Hysterectomy; Infertility; Intravenous administration; Magnetic resonance imaging; Malignancy; Medical imaging; Menstrual cycle; Movement disorders; Multiple cerebral infarctions; NMR; Nuclear magnetic resonance; Ovarian cancer; Recurrence (Disease); Tumor markers; Uterus; Warfarin; Women
• ISSN: 1471-2377; 1471-2377
• DOI: 10.1186/s12883-018-1117-1

Benign gynecologic tumor, such as uterine adenomyosis, has been suggested to develop hypercoagulability. Although some cases of cerebral infarction associated with adenomyosis have been reported, the mechanism of hypercoagulation initiated by adenomyosis is still not clear, and the therapeutic strategy is uncertain. A 44-year-old woman was presented to our department with headache, left hand weakness, and gait disturbance during her menstrual phase. She had a history of adenomyosis and infertility treatment for 18 years and heavy menstrual bleeding. Magnetic resonance imaging on admission showed multiple hyperintense lesions in cortical and subcortical areas in the cerebrum and cerebellum on diffusion-weighted imaging. Transesophageal echocardiography showed neither embolic sources nor existence of foramen ovale. Her laboratory data revealed anemia, a high D-dimer level, and elevated levels of a mucinous tumor marker. She had adenomyosis and no malignancy was detected. Anticoagulation therapy with intravenous heparin followed by rivaroxaban did not prevent recurrence of cerebral infarction. We discontinued rivaroxaban, and started warfarin therapy with pseudomenopause treatment, which prevented recurrence for 6 months. Five months after her last pseudomenopause treatment, multiple cerebral infarctions occurred. Total hysterectomy was performed, which prevented recurrence of the multiple cerebral infarctions for 2 years without anticoagulation therapy. Our findings reveal for the first time that anticoagulation therapy, including novel oral anticoagulants, had no preventive effect against cerebral infarctions associated with adenomyosis in a middle-aged woman. Although pseudomenopause treatment temporarily prevented recurrence, resection of the adenomyosis might be the most effective therapy in these cases.