Lamotrigine (LTG) has been used in epilepsia patients for treatment of partial seizures. It can cross the placenta and there are limited data about its use in pregnancy and foetal adverse effects. Extremity and cardiac malformations, dysmorphic facial appearance, coanal atresia and upper respiratory and gastrointestinal anomalies have been reported because of LTG use in pregnancy. Oxcarbazepine (OXC) is one of the new antiepileptic agents. Although the drug and its metabolites can easily pass from placenta to the foetus, available data suggest that it can be safely used during pregnancy because no teratogenicity has been reported. One infant with atrial septal defect and patent ductus arteriosus due to use of LTG and OXC in pregnancy has been reported in literature. Here, we report a female infant with micrognatia, low-set ears, facial dysmorphism and unilateral radius aplasia born to a mother who used LTG 100 mg/day and OXC 1200 mg/day during pregnancy for seizures. To our knowledge, this is the first major anomaly case associated with the combined use of these drugs. This case can provide useful data about the teratogenicity of LTG and OXC combination therapy.