We report a case of a 21-year-old woman who underwent radiosurgical treatment of a distal lenticulostriate artery (LSA) aneurysm. Twenty-two months after treatment, repeat angiography demonstrated patency of the parent vessel and complete obliteration of the aneurysm. Our case implies that Gamma Knife surgery (GKS) might serve as an alternative microinvasive technique in the treatment of LSA aneurysms, making this procedure a potential addition to present methods.