Endogenous Aspergillus endophthalmitis (AE) is a rare complication of invasive aspergillosis (IA) in transplant patients. In this report, we describe a patient who underwent liver transplantation because of drug-induced cholestatic cirrhosis who developed AE 2 weeks after the surgery. The patient was a 22 y/o male who received right lobe of the liver from his father. The operation was uneventful but the patient developed signs and symptoms of small-for-size syndrome after second day of the surgery. The patient received intense immunosuppression with methylprednisolone for 3 days, tacrolimus and mycophenolate mofetil (MMF) from the first day after the operation and ceftriaxone and metronidazole as prophylactic antibiotics. Because of signs of respiratory distress with pneumonia vancomycin and amphotericin B added empirically to his regimen. PCR for aspergillus DNA in the blood was positive. The patient received 1 course of methylprednisolone pulse therapy for signs of acute rejection at 10th day and tacrolimus changed to sirolimus because of rising serum creatinine and convulsion. After 2 weeks the patient’s symptoms improved and liver function tests were normal but the patients complained from sudden intense pain in the left eye with unilateral blurred vision, redness and other signs of endophthalmitis in the eye examination by ophthalmologists. Visual acuity decreased to light perception after 24 hours. AE was confirmed by microscopy and culture of the vitreous fluid and retinal biopsy. Despite changing amphotericin to intravitreal injection of voriconazole followed by intravenous voriconazole and transient resolving of the symptoms, no improvement was seen in visual acuity, and pain and signs of inflammation of the eye recurred after 2 weeks. At last the patient underwent enucleation for resistant infection and fear of involvement of the other eye by aspergillosis or sympathetic ophthalmia.