We report two patients with complete callosal agenesis in whom Probst bundles in both hemispheres could be depicted by diffusion tensor tractography (DTT). While one patient had no associated telencephalic anomaly other than callosal agenesis, the other had cortical dysplasia in the right frontal lobe. Although Probst bundles in the three normal hemispheres were well developed, that in the hemisphere which was affected by cortical dysplasia was small and poorly developed. DTT also showed that the fibers from the frontal pole ran more on the inner side of the Probst bundle than those from a more caudal region of the frontal lobe. Furthermore, fibers from the orbital gyri ran along the outermost side of Probst bundle. The arrangement of these fiber tracts in Probst bundle may reflect the developmental process of callosal fibers in their normal formation.