Hemolytic uremic syndrome (HUS) is a common form of thrombotic microangiopathies. Among its extrarenal complications, ocular involvement is very rare. We present a patient with HUS, whose first symptom was isolated abduction deficits in the eyes. Lethargy was added during the clinical course, suggesting neurological involvement. Although conventional magnetic resonance imaging was normal, symmetric diffusion restriction was detected in bilateral putamen on diffusion-weighted images. Treatment with peritoneal dialysis, fresh frozen plasma infusions, and eculizumab was initiated. The patient responded well to the treatment and was discharged with excellent neurological, hematological, and ophthalmological outcomes. Nephrological follow-up is being continued due to proteinuria To our knowledge, presenting with ophthalmoplegia in the acute phase of hemolytic uremic syndrome is very rare. The patient’s ophthalmological and neurological symptoms improved after eculizumab treatment. We suggest that eculizumab is effective in the acute phase of HUS in cases of ophthalmological involvement.

Keywords: Ophthalmoplegia, hemolytic uremic syndrome, diffusion restriction, putamen, eculizumab