Résumé : Infective endocarditis (IE) due to non-HACEK (species other than Hemophilus, Aggregatibacter, Cardiobacterium, Eikenella, Kingella) bacteremia accounts for less than 2% of all IE cases but is proven to be associated with higher mortality, even more so in hemodialysis (HD) patients. Few data are available in the literature concerning non-HACEK Gram-negative (GN) IE in this immunocompromised population with multiple comorbidities. We report the atypical clinical presentation of an elderly HD patient diagnosed with a non-HACEK GN IE, namely E. coli, successfully treated with intravenous (IV) antibiotics. The objective of this case study and related literature was to highlight the limited applicability of the modified Duke criteria in the HD population, as well as the frailty of HD patients that increases their susceptibility to IE due to unexpected microorganisms that could have fatal consequences. The need for a multidisciplinary approach of an IE in HD patients is therefore imperative.