Background Cerebral Hyperperfusion Syndrome (CHS) is an uncommon complication observed after intracranial angioplasty or stenting procedures. Given to the increasing use of new devices for intracranial angioplasty and stenting (INCS), in selected patients with high ischemic stroke risk, an equally increasing knowledge of complications related to these procedures is mandatory. Case description: a 63-year-old man was diagnosed with an hyperperfusion syndrome after percutaneous angioplasty and stenting for severe symptomatic right internal carotid artery (ICA) siphon stenosis. After treatment he complained generalized seizures and respiratory failure. While conventional imaging did not demonstrate any acute brain lesions, Pseudo-Continuous Arterial Spin Labeling (PCASL) Perfusion MRI early documented right hemisphere blood flow increase suggestive for CHS. Conclusions Monitoring of perfusion changes after INCS could play an important a role in determining patients with high risk of CHS. ASL Perfusion MRI might be used for promptly, early diagnosis of CHS after treatment of severe intracranial artery stenosis.