Aim:The cerebral aqueduct is a deep structure located in the diencephalon. It is part of the ventricular system and connects the third and fourth ventricles. The aqueduct has three portions: superior, medial, and inferior. Pure aqueduct tumors occur in the superior portion, and lesions located within the aqueduct are difficult to remove through conventional approaches. Most aqueduct tumors are biopsied and accompanying hydrocephalus is addressed through an endoscopic third ventriculostomy (ETV). The extreme anterior interhemispheric transcallosal approach makes use of a natural corridor to remove aqueduct lesions. Herein, we describe the detailed anatomy of this approach for resecting pure aqueduct tumors in the superior portion of the aqueduct. We aimed to show the normal anatomy of the cerebral aqueduct and the feasibility of the approach to remove tumors within the aqueduct. Material and Methods:Ten human brain hemispheres and one cadaveric head were dissected under an operative microscope with 6x to 40x magnification. The cerebral aqueduct anatomy was delineated along with the relationship to nearby structures in the extreme anterior interhemispheric transcallosal approach. Results:We described the anatomy of the cerebral aqueduct within the brain and showed that, with the proper angle for the extreme anterior interhemispheric transcallosal approach, lesions in the cerebral aqueduct can be reached in a single session without damaging periventricular structures.Conclusion:The extreme anterior interhemispheric transcallosal approach provides a direct corridor to the cerebral aqueduct and, thus, is feasible for resecting pure aqueduct tumors in an already dilated intraventricular foramen.