Lenticulostriate artery (LSA) aneurysms are uncommon. Most reported cases have been surgically treated. There are only three reports of LSA aneurysms treated with endovascular techniques. We report the case of a 49-year-old man who presented with a hemorrhage in the basal ganglia. An angiogram revealed a distal LSA aneurysm in the setting of moyamoya disease. The patient's aneurysm was successfully embolized by the use of Onyx 18. In this report, we present the first case of onyx embolization of a LSA aneurysm and review all the previous cases that were managed with endovascular techniques. Treatment of these lesions with either surgery or endovascular therapy is challenging and requires specialized expertise. Given the risks of surgery, we believe that LSA aneurysms are best treated by endovascular means, especially in the setting of moyamoya disease.