Abstract
Linear nevus sebaceous syndrome (LNSS) is a rare neurocutaneous syndrome with important neurological involvement including brain malformation, focal seizures, and developmental delay. We discuss a case with a unique presentation with localization-related infantile spasms and review the clinical and radiological features of this case. To our knowledge, there are no previously reported cases of LNSS with infantile spasms and cortical dysplasia. Therefore, the presented case will make an important contribution to the available knowledge.