Cor triatriatum sinistrum (CTS) is a congenital anomaly where the left atrium is divided into two compartments by a fibromuscular membrane. This report aims to add to the literature on a rare cardiac condition that can cause neurological morbidity. We report a case of a 19-year-old female with an infarct in the right middle cerebral artery (MCA) territory initially maintained on aspirin. Eighteen months later, she had recurrence of weakness, for which repeat transthoracic echocardiography (TTE) and re-evaluation of the first TTE demonstrated a hyperechoic membrane spanning the width of the left atrium, clinching the diagnosis of CTS. Despite anticoagulation with apixaban, she was admitted for a third stroke where she succumbed to hospital-acquired pneumonia. Among cases of CTS associated with stroke, anticoagulation and surgery were the main modes of treatment. This case has the longest follow-up and the first to demonstrate failure of antiplatelet therapy and anticoagulation.
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