Strabismus is a common eye disease characterized by abnormal eye position and ocular motor disorder. In this study, we compared the cerebral blood flow (CBF) in patients with comitant extropia (CE) relative to healthy controls using 3D-pCASL MRI. We found that CE patients had significantly increased CBF in the right parahippocampal region, bilateral medial FG/ACC, bilateral IFG, left SFG, bilateral MCC, right MFG (BA8), and right paracentral lobule. This study demonstrates the hypothesis that CE involves the dysfunction of visual pathway. Interestingly, the most significant CBF increase in the right parahippocampal region, suggests potential cognitive and mood compensation in CE.