Early detection of focal cortical dysplasia (FCD) using brain MRI in young children presenting with drug-resistant epilepsy may facilitate prompt surgical treatment, resulting in better control of seizures and decreased associated cognitive difficulties. Characteristics of FCD described in the literature are predominantly based on MRI findings in a fully myelinated brain; therefore, changes occurring during early brain maturation are not well known. In this case report, we describe distinct MRI features of a FCD visualized best before completion of myelination of the cortex and subcortical white matter. Specifically, this lesion exhibited increased visibility because of better delineation of the borders of the pathological area involved in the malformation compared with imaging obtained after completion of myelination. Although the pathophysiology of these imaging features necessitates further investigation, one hypothesis is that excessive neuronal input in young epileptic patients could trigger premature myelination of overstimulated fibers underlying the cortical epileptogenic focus. The aim of this case report was to raise awareness that seizures might induce local changes in brain myelination, which can be detected by MRI during the first year of life. Early identification of FCD may prompt surgical treatment in selected patients with drug-resistant epilepsy.