PurposeTo describe observations by ultrahigh-resolution optical coherence tomography (OCT) in a secondary corneal amyloidosis (SCA) patient with histological analysis of excised tissue. A unique finding under OCT of her fellow eye is also described.CaseA 39-year-old female had suffered from trichiasis in both of her eyes for more than 30 years. Slit-lamp examination showed a milky-white soft mass on her left cornea and a linear opacity on the fellow cornea at the cilia-attached region. OCT demonstrated the presence of a mass region within a thin epithelial layer and no destruction of Bowman’s layer in her left cornea. In the fellow cornea, which exhibited a linear opacity, a high-density spot in Bowman’s layer was observed at the cilia-attached region covered by the epithelial layer, with normal thickness. Histological examination of the excised cornea showed that the mass was positive with both Congo red and antilactoferrin antibody.ConclusionSCA, amyloid gradually accumulates above Bowman’s layer, occupying the epithelial layer, with no destruction of Bowman’s layer until the advanced stage. A high-density spot in Bowman’s layer might be the first stage of SCA.