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Atypical hemolytic uremic syndrome (aHUS) represents a major challenge due to its rare nature and severe impact on patients, characterized by microangiopathic hemolytic anemia, thrombocytopenia, and acute renal failure. This paper describes the case of a 41-year-old patient diagnosed with aHUS in the context of malignant arterial hypertension and severe renal impairment, manifested by anuria and significant azotemic retention syndrome. The complex management of the case, including the use of Eculizumab, a complement inhibitor, highlighted significant therapeutic benefits, especially in improving hematological parameters. Despite a positive response, challenges related to dosing, monitoring treatment efficacy, and maintaining remission without relapses emphasize the need for adapted therapeutic strategies and a deeper understanding of the disease mechanisms. This case also highlights the importance of individualized approaches and consideration of the possible benefits of dose adjustments based on specific clinical and pharmacological parameters, as well as continuous evaluation of treatment efficacy and safety, in the context of such a variable and potentially devastating syndrome as aHUS. [ABSTRACT FROM AUTHOR]