Eculizumab has proven to be effective in patients with atypical hemolytic uremic syndrome (aHUS) in clinical trials and in the real world, but the optimal duration of therapy remains unknown. Standard maintenance treatment is often life-long, but the possibility of discontinuation has not yet been systematically tested. We describe a case of aHUS after ChAdOx1 nCoV-19 vaccination in a patient with homozygous CFHR3/CFHR1 gene deletion who discontinued eculizumab maintenance therapy 24 weeks after achieving disease remission. We report the safety of discontinuing eculizumab treatment with the aim of minimizing the risk of adverse reactions, reducing the risk of meningitis, improving quality of life, and reducing the considerable treatment costs.