The corpus callosum develops from anterior to posterior, starting at t
he commissural plate of the lamina terminalis. We report a patient wit
h cortical dysplasia and schizencephaly, which apparently interfered w
ith the normal callosal development before 20 gestational weeks. The r
esult was an atypical callosal dysgenesis in which the anterior parts
(including anterior body, genu and rostrum) were absent while the rema
ining parts were developed. This finding suggests that the commissural
plate may not be the only region where the corpus callosum starts to
develop in some congenital brain malformations.