Anticonvulsant drugs taken by pregnant women to prevent seizures are among the most common causes of potential harm to the fetus. It has been suggested that carbamazepine was less teratogenic than the other drugs. Here, we report a case of fetal carbamazepine syndrome presenting with facial dysmorphism, congenital heart defect, skeletal abnormalities, renal agenesis, ambiguous genitalia, anal atresia, and right hemihypoplasia of the entire body. To the best of our knowledge this is the most severe case of fetal carbamazepine syndrome in the literature. This case can provide useful data about teratogenicity of carbamazepine therapy during the pregnancy.
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