The aim of this work was to assess the structural and functional state of the retina in a murine model of ALS caused by overexpression of the aberrant FUS protein [1-359]. The retinal examination was carried out on 12 transgenic and 13 wild-type mice of 2.5-3 months of age. The study revealed not statistically significant higher level of ophthalmoscopic violations in FUS[1-359] mic