Objective: Lenticulostriate artery (LSA) aneurysms are rare. We present 2 cases of LSA aneurysm, their clinical presentation, diagnosis, surgical treatment, and postoperative follow-up, with a review about the topic.Case Report: We report 2 female cases of LSA aneurysms among 194 surgically treated aneurysms in Santa Paula and Sa˜o Camilo Hospital, who underwent the same surgical treatment. The first patient is a 48-year-old, Latin American woman whose aneurysms were detected incidentally in an angiographic study performed owing to a cavernous sinus thrombosis. Both cerebral angiogram and magnetic angioresonance were performed and demonstrated a left lenticulostriate aneurysm and 2 parassylvian aneurysms. The other patient is a 62-year-old, Japanese, hypertensive patient who presented with subarachnoid hemorrhage, Fisher scale 2, and Hunt-Hess classification 2. Her angiogram demonstrated a right LSA, left middle cerebral artery, and anterior communicating artery aneurysms. No other pathology or infectious etiology was noted. Both patients were treated by opening of the sylvian fissure, allowing the visualization of lenticulostriate vessels, and aneurysm clipping. Postoperative angiographies were performed in both the cases. During 2-year follow-up the younger patient remained just with distal right arm paresis and Rankin scale score 1; the older patient developed normal pressure hydrocephalus, needing peritoneal ventricular drainage.Conclusions: LSA aneurysms are uncommon. Subarachnoid hemorrhage and intracranial sinus thrombosis are quite more common pathologies nevertheless the underlying vascular abnormality is rarely identified. The most common clinical presentation is intraparenchymal hemorrhage. Microsurgical treatment is often the chosen modality of intervention. Elderly people are more likely to develop postoperative complications before and after hospital discharge, which may be associated to microscopic alterations in vessels. They are also under higher risk of more severe vasospasm and risk of hydrocephalus development in cases of ruptured aneurysms.