AIM:To report a male patient with Pickwickian syndrome and obstructive sleep apnea(OSA)who presented with pseudotumor cerebri and visual loss.METHODS:Case report.RESULES:A 54-year-old obese man with a three-month history of bilateral visual deterioration was evaluated.His visual acuity was 20/200 in OD and 20/400 in OS.Color vision was impaired only in the left eye.Funduscopy revealed bilateral disc edema and peripapillary hemorrhages together with macular exudates in OS.Physical examination and laboratory investigations were consistent with OSA and Pickwickian syndrome.Lumbar puncture demonstrated an opening pressure of 350mm H2O and computed brain scan was normal.The diagnosis was pseudotumor cerebri in association with OSA and Pickwickian syndrome.Fundus findings and visual acuity improved with phlebotomy,blood pressure regulation,weight reduction and bi-level positive airway pressure therapy.CONCLUSION:OSA and Pickwickian syndrome should be kept in mind when facing a patient with pseudotumor cerebri.