A case of agenesis of the internal carotid artery with ipsilateral aneurysm of the anterior middle cerebral artery is first described in the literature. This is a 23-year-old male with seizures in whom computed tomography and magnetic resonance imaging (MRI) revealed a large left paracellular calcified formation. MRI showed no ipsilateral aneurysms of the internal carotid artery and carotid canal. Agenesis was confirmed by intraoperative data at removal of thrombotic aneurysm. The paper provides a brief account of the outcome of surgery and a review of literature on agenesis of the internal carotid artery concurrent with cerebral aneurysms.
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