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Neurotoxicity is a rare and intolerable adverse effect of ceftriaxone therapy. In most cases, it has been diagnosed on the basis of medical history rather than quantitative blood and cerebrospinal fluid testing. We report the case of a woman aged 78 years with ceftriaxone-associated encephalopathy. She regularly underwent hemodialysis. The patient received intravenous ceftriaxone at a dose of 1 g/day for 10 days for a urinary tract infection, and her consciousness level began to deteriorate during the therapy. Five days after ceftriaxone discontinuation, her symptoms rapidly improved. Thus, ceftriaxone-associated encephalopathy was suspected. Ceftriaxone levels in the blood and cerebrospinal fluid were high while the patient had disturbed consciousness. This case showed that ceftriaxone levels were related to ceftriaxone-associated encephalopathy. Therefore, the estimation of ceftriaxone levels may facilitate an accurate diagnosis., (Copyright © 2022 The Author(s). Published by Elsevier Ltd.. All rights reserved.)