Abstract. Intravitreal bevacizumab injection (IVB) is emerging as a safe and effective therapy for retinopathy of prematurity (ROP); however, follow-up investigations after IVB have indicated that endophthalmitis, a rare and devastating complication, may develop. The present study reports a case of an infant with ROP who developed endophthalmitis after IVB infection. The infant was administered with an intravitreal injection of broad-spectrum antibiotics to treat the endophthalmitis. A favorable anatomic outcome was achieved after follow-up. From this case, it was discovered that the early clinical characteristics of endophthalmitis infection secondary to IVB in premature infants include: i) The clinical symptoms of endophthalmitis infection appearing as early as 3-5 days after IVB; ii) a white membranous plaque on the retina surface with an obscure circular boundary; iii) a plaque that enlarges to a gray flocculent mass and intrudes the vitreous body, where the hazing surrounds the lesion; and iv) conjunctival hyperemia and cornea edema in the anterior segment of the affected eye. This case also indicated that timely retinal screening with RetCam or a binocular ophthalmoscope 3-5 days after IVB may be effective for detecting early stage endophthalmitis in infants who are unable to complain of a loss of visual acuity. From the present case report, it is advisable that the same type of screening be performed 3-5 days after other types of eye surgery, such as cataract extraction surgery, to detect early stage endophthalmitis. Furthermore, the present case also revealed that an early intravitreal injection of broad-spectrum antibiotics may be an effective treatment for premature infants with bacterial endophthalmitis.