Cerebral infarction associated with benign mucin-producing adenomyosis: report of two cases

BACKGROUND: Cerebral infarction associated with a malignant tumor is widely recognized as Trousseau syndrome. In contrast, few cases of cerebral infarction associated with benign tumors have been reported. We present two cases of embolic stroke that seemed to be caused by mucin-producing adenomyosis. CASE PRESENTATION: The patients were women aged 42 and 50 years old. Both patients developed right hemiparesis and aphasia, and cerebral infarctions were detected in the left cerebral hemisphere. There were no other abnormal findings, except for elevation of CA125 and D-dimer. Trousseau syndrome was suspected in both cases, but whole body examinations did not reveal any malignant tumors. However, uterine adenomyosis was detected in both patients. CONCLUSIONS: From our findings and a review of the literature, both mucin-producing malignant tumors and mucin-producing benign tumors such as adenomyosis may cause hypercoagulability and cerebral infarction. This mechanism should be considered in a case of a young to middle-aged woman with embolic stroke of an undetermined origin.