Objective Lenticulostriate artery (LSA) aneurysms are rare. Here we present 2 cases of LSA aneurysm, their clinical presentation, diagnosis, surgical treatment, and postoperative follow-up, with a review of the topic. Case Report We report 2 female patients with LSA aneurysms among 194 surgically treated aneurysms in the Santa Paula and São Camilo Hospitals, who underwent the same surgical treatment. The first patient was a 48-year-old, Latin-American woman whose aneurysms were detected incidentally in an angiographic study carried out due to a cavernous sinus thrombosis. Both cerebral angiogram and magnetic angioresonance were performed and showed a left lenticulostriate aneurysm and 2 parasylvian aneurysms. The other case was a 62-year-old, Japanese, hypertensive patient who presented with subarachnoid hemorrhage, Fisher scale 2, and Hunt-Hess classification 2. Her angiogram showed 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 the sylvian fissure, allowing the visualization of the lenticulostriate vessels and aneurysm clipping. Postoperative angiographies were performed in both cases. During the 2-year follow-up the younger patient remained with just distal right arm paresis and Rankin scale score of 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 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 with microscopic alterations in vessels. They are also at higher risk of more severe vasospasm and the development of hydrocephalus in cases of ruptured aneurysms.