The genus Dermocystidium is very comprehensive in the host and site of infection, however this is the first report of the occurrence of Dermocystidium sp. in the gills of Nile tilapia. This study was carried out in a fish farming located in the state of Santa Catarina, Brazil. No mortalities were reported in the facility studied and the animals were clinically healthy. During the histopathological analysis of the gills, 8.33% of the fish presented spores of Dermocystidium sp. in the gill tissue. The spores reported herein had a mean length and width of 6.206 x 5.233 μm and a refractile body diameter of 1.965 μm and were studied by histopathology and Transmission Electron Microscopy. This study highlights the importance of a new branchial pathogen in farmed tilapia, as well as to its pathogenic potential, considering the outbreaks of mortalities associated with other fish species.