| Summary: | Key Clinical Message We reported herein a case of isolated cerebral actinomycosis in a 54‐year‐old immunocompetent man. Brain MRI showed a left frontal intra‐axial lesion and perilesional edema. We performed an open biopsy of the left frontal enhancing lesion. Intraoperative findings showed a yellowish, malleable, and capsulated lesion that was well defined with surrounding normal tissue within pus inside and lacked any necrotic content. MR spectroscopy showed a high level of choline, lactate, and lipid peaks with a choline/N‐Acetylaspartic acid ratio of 1.8. The diagnosis was confirmed histologically, and the patient was treated successfully for 3 months after surgical aspiration. Surgical management allowed to confirm the diagnosis with a shorten antibiotics, a rapid resolution of symptoms, and a complete recovery.
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