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Objective: The optimal treatment of in-stent restenosis (ISR) is not yet determined.We evaluated carotid endarterectomy (CEA) with stent removal in management of ISR.Methods: We described a new case and performed a search of English literature in PubMed and Google Scholar.Keywords used were (single word or combination): " carotid endarterectomy"; "carotid artery stenting"; "in-stent restenosis"; "restenosis"; " recurrent stenosis".Only patients treated by CEA with stent removal due to ISR were included.Results: A 72-year-old man presented with left internal carotid artery ISR 8 months after carotid artery stenting.CEA with stent removal was performed.40 cases meet the above criteria were found.Including our patient, a total of 41 cases were ana lyzed.About 10.3% showed intense periarterial in?ammation.92.9% had clear intima media plane.Patch angioplasty was used in 80.5% of all cases.7.3% had graft inter position.Postoperatively, 85.4% had a good outcome, 7.3% presented with transient ischemic stroke, and 7.3% suffered neck hematoma.During a mean follow-up of 15.3 months, all patients remained stable and without recurrent restenosis.Conclusions: CEA with stent removal appears to be safe, feasible, effective and durable therapeutic option.Due to the limitations of the currently available data, ran domized controlled trials are highly recommended.