Introduction: Anterior cingulate epilepsy is a peculiar epileptic syndrome with a broad range of clinical manifestations, depending on the numerous projections of anterior cingulate into motor systems. Its diagnosis is often delayed, as seizures mostly occur during sleep and are typically misdiagnosed as parasomnias. Moreover, most focal anterior cingulate epilepsies are believed to be cryptogenic or idiopathic, even if there are some reports of anterior cingulate cortical dysplasia, while anterior cingulate neoplasms underlying epilepsy are rare.
Case report: Here, we report a 30-month-old boy who developed, at the age of 20 months, cingulate epilepsy associated with a low-grade oligodendroglial tumor. It must outlined that the clinical presentation was characterized by very frequent and disabling seizures as the only symptom of the disease, while the results of several neuropsychological tests suggested intact intellectual and behavioral abilities.
Discussion and conclusion: Seizures disappeared completely after surgical removal of the lesion and neuropsychological child's performances remained completely normal. Long-term follow-up and observation are essential for evaluating the future clinical course.