Purpose: To report optical coherence tomographic (OCT) findings in Susac syndrome. Methods: A 15-year-old girl who had symptoms of deafness and encephalopathy was referred with a provisional diagnosis of Susac syndrome. Funduscopy revealed cotton wool spots and ischaemic cloudy swelling of the retina. Results: OCT taken from the abnormal retinal area showed increased reflectivity of the inner retinal layers and decreased reflectivity from the outer retinal layers and the retinal pigment epithelium due to shadowing. Neither intraretinal nor subretinal fluid were observed on the OCT imaging. OCT findings were compatible with the accepted view that the retinal pathology in Susac syndrome is due to arteriolar occlusion. Conclusion: OCT may be used as a complementary diagnostic method to fluorescein angiography in Susac syndrome.