Fatal infection by Cyathostoma (Cyathostoma) phenisci (Nematoda: Syngamidae), was identified in 2 of 52 brown boobies (Sula leucogaster) collected on beaches in the state of Rio de Janeiro, Brazil, and admitted to the veterinary clinic for rehabilitation. Both infected birds were in poor physical condition, with atrophied pectoral muscles, and died soon after starting treatment. The parasitological and pathological examination of the carcasses revealed the presence of C. (C.) phenisci in the trachea, resulting in tracheitis, as well as severe parasitic granulomatous bronchopneumonia caused by eggs deposited in the lungs. In our opinion, these serious pathological changes were the primary cause of chronic respiratory illness. This is the first description of fatal cyathostomiasis in a fish-eating avian host caused by infection by a member of the subgenus Cyathostoma (Cyathostoma). Therefore, it is reasonable to consider C. (C.) phenisci to be a real threat to a wide range of their definitive hosts, and cyathostomiasis should be considered in the differential diagnosis for fish-eating marine birds, even in cases without respiratory signs. This is also the first record of the genus Cyathostoma in Brazil.