The first ophthalmosaurid ichthyosaur from the Upper Jurassic deposits of the Central–Northern Apennines (Marche, Italy) is here described for the first time. The specimen is relatively complete and is referred to Gengasaurus nicosiai gen. et sp. nov. based on a unique combination of characters, including a peculiar condition of the preaxial accessory facet on the humerus. The faunal association of the ichthyosaur-bearing level indicates a late Kimmeridgian – earliest Tithonian age, and its finding contributes significantly to our knowledge of the diversity of Late Jurassic ichthyosaurs from the Western Tethys. Two shark teeth assigned to the order Hexanchiformes were also recovered in association with the ichthyosaur specimen, suggesting that scavenging of the carcass might have occurred. Gengasaurus can be referred to Ophthalmosauridae based on the reduced extracondylar area of the basioccipital, and the presence of a preaxial digit. It differs from Ophthalmosaurus spp. in several respects, including the shape of the posterior basisphenoid, the shape of the supraoccipital, the anteriorly deflected preaxial facet of the humerus, and a proximodistally shortened ulna. The new taxon actually shares diagnostic characters with both members of the two main lineages recovered in previous phylogenetic analyses, more nested within Ophthalmosauridae. The affinities of Gengasaurus to genera from both the northern and southern hemispheres also suggest that connectivity between pelagic habitats was high during the early Late Jurassic, allowing dispersal of some forms, followed by local, endemic divergence.