Levetiracetam is a relatively novel antiepileptic drug used for the treatment of partial and generalized seizures in adult and children. Several animal studies describe a possible drug adverse effect on skeletal growth and metabolism. We present a case report of a 10-year-old female child who underwent a prolonged 7-year treatment with levetiracetam for sporadic secondary convulsions secondary to enterovirus encephalitis at the age of 15 months. This patient developed an osteochondritis dissecans lesion (OCD) of the talar head well treated conservatively. Only a few cases have been described of this rare type of OCD. We hypothesize a possible association between levetiracetam therapy and OCD development, suggesting the importance of long-term control of bone growth in levetiracetam-treated pediatric populations.