.....A previous study has found that ex vivo Aβ amyloid plaques in the retina of a transgenic mouse model appear hyper-autofluorescent (hyper-AF) under specific wavelengths (excitation at 830 nm, emission at 630). We report the first description of hyper-AF ocular findings in a case of transthyretin-related familial amyloid polyneuropathy (FAP) associated with ocular involvement using AF imaging. A 47-year-old woman from Italy presented to our institution with a history of early onset FAP with ocular involvement due to the rare amyloidogenic transthyretin Glu54Lys mutation. AF imaging showed hyper-AF amyloid deposits associated with retinal vessels in both eyes and a hyper-AF amyloid deposit anterior to the optic disk in the right eye. Conclusion and Relevance: Further studies in FAP patients with different types of genetic mutations and various disease-stages are needed to better establish if non-invasive AF imaging is useful in detecting ocular amyloidosis.