Vasospasm following aneurysmal subarachnoid hemorrhage (SAH) occurs in the extraparenchymal vessels in the subarachnoid space at the base of the brain. Ischemia/ Infarction affecting primarily the perforator vessels in isolation, following aneurysmal SAH is uncommon. A 28-year-old man with a ruptured middle cerebral artery aneurysm underwent clipping of the aneurysm. He developed delayed bilateral deep seated infarcts involving both internal capsular regions, the thalamus and basal ganglia without any major vessel infarct. The patient was managed with triple H (hypertensive hypervolemic hemodilutional) therapy and calcium channel antagonists but did not show any improvement and remained in poor neurological status. Perforator vasospasm occurring secondary to aneurysmal SAH, though documented in experimental animal studies, has rarely been reported in humans in a clinical setting. The present case provides evidence, albeit indirect, of isolated perforator vasospasm, which possibly should be the target of future therapeutic strategies.