Hypoxia during sleep in obstructive sleep apnea syndrome (OSAS) increases intracranial pressure, decreases cerebral perfusion pressure, and alters vascular supply to the optic nerve. Pattern visual evoked potential (pVEP) has revealed that it causes alterations in the optic nerve, and optic coherence tomography has shown that it causes alterations in the retinal and macular layers. To detect and compare possible alterations in macula and peripapillary retinal nerve fiber thickness (pRNFL) using OCT and in the optic nerve pathways using pVEP before and after positive airway pressure (PAP) in the patients with severe OSAS. Thirty patients who were diagnosed as having severe OSAS in the neurology-sleep outpatient clinic and 30 healthy control subjects were included in the study. Ophthalmic examinations were performed prior to (month 0) and after (month 6) PAP treatment, and pVEP (peak time [PT] and amplitude) and OCT parameters (peripapillary retinal-macular layers) were compared. In the comparison between the severe OSAS (before treatment) and control groups, thinning was found in pRNFL (average, nasal, inferior) and in the macular layers (external and internal superior quadrants) (p < 0.05). pVEP investigation revealed increased PT in P100 and N145 waves and decreased amplitude of N75-P100 waves. In the comparisons before and after PAP treatment, a decrease in PT of N75 and P100 waves and increase in N75-P100 amplitudes were found. In the pRNFL, significant thickening was found in the layers with thinning before treatment, whereas no significant thickening was found in macular layers, except for the fovea. It was shown that PAP treatment in patients with severe OSAS prevents hypoxia without causing alterations in intraocular pressure and thus reduces inflammation and causes thickening in the pRNFL and macular layers.