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Cefepime is a fourth-generation cephalosporin utilized in treatment of multiple Gram-negative and -positive infections. The current report presents a case of 50-year-old man admitted with epidural abscess who developed neutropenia after prolonged use of cefepime. The neutropenia developed after 24 days of cefepime treatment and resolved 4 days after cessation of cefepime. Assessment of the patient's profile indicated no other possible cause for neutropenia. A literature review was done, and is presented herein to compare and identify the pattern of cefepime-induced neutropenia in 15 patients. The data presented in this article highlight that despite its rarity, cefepime-induced neutropenia should be considered by clinicians when planning a prolonged course of cefepime., Competing Interests: The authors report no conflicts of interest in this work., (© 2023 Koraysh et al.)