ObjectiveTo analyse the seizure semeiology of MRI negative insular epilepsy.MethodsA case of patient with insular epilepsy who presented in Epileptic Center, Guangdong 999 Brain Hospital was collected. Related literatures were thoroughly reviewed.ResultsThe patient was diagnosised as insular epilepsy by SEEG and postoperative pathology reported focal cortical dysplasia Ib.ConclusionsThe insula lobe is a part of the limbic system, and the seizure semeiology in insular epilepsy is varied. Seizure semeiology analysis is particularly important in the evaluation of presurgical especially in a MRI negative case. In this paper, we summarized the semeiology characteristics of various parts of insula lobe, and provided some references for the diagnosis and treatment of insular epilepsy.